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test-301
5721577
{'Case report': "The patient was a 37-year-old female, of 157 cm in height and 45 kg in weight. She was transferred to our department from a local hospital due to psychiatric features (both agitated behavior and auditory hallucinations), alexia and apraxia that had begun 10 days ago, followed by disorientation and generalized tonic-clonic seizures. She had a long history of episodic migraine-like headaches and progressive bilateral hearing loss for 3 years. However, she did not take any medication. Her vital signs showed a normal body temperature of 36.8 °C, a hypotension of 90/56 mm Hg and pulse at 72 beats per minute. Because of the patient's psychiatric symptoms and application of sedative after seizure attacks, she could not cooperate with physical examination. The laboratory data showed high anion gap metabolic acidosis with elevated levels of lactate and pyruvate. Serum levels of thyroid-stimulating hormone (TSH) and free thyroxine (FT4) were decreased. Her TSH level was low at 0.26 mU/L (normal range 0.35–5.5 mU/L), and FT4 concentration was 7.56 pmol/L (normal range 10.2–31 pmol/L). Both serum free and total triiodothyronine (FT3 and TT3) were significantly lower than normal range. FT3 concentration was 2.28 pmol/L (normal range 3.5–6.5 pmol/L), and TT3 concentration was 0.6 nmol/L (normal range 1.2–3.4 nmol/L). Moreover, elevated titers of serum anti-thyroglobulin and anti-thyroid microsomal antibodies were detected. Cerebral spinal fluid (CSF) studies were normal for cell counts and biochemistry, and negative for culture. CSF IgG index was 0.48 (normal range ≤ 0.7), and oligoclonal bands (OB) was negative. Computed tomography (CT) scan showed lesions of hypodense in the left temporal and parietal lobe, with brainstem and cerebellar atrophy ( Fig. 1 A). No evidence of subarachnoid or intracerebral hemorrhage. Vascular imaging of the cervical and cerebral arteries by CT angiography excluded the possibility of cerebrovascular disease ( Fig. 1 B). However, CT images were not conclusive to differentiate between the infectious or metabolic lesion. On day 5, brain magnetic resonance imaging MRI (3.0 T) revealed a hypointensity lesion in the left temporal-parietal lobe on T1 weighted image ( Fig. 2 A), and increased signal intensity in the same region on FLAIR sequences ( Fig. 2 B) with a clearly restricted diffusion ( Fig. 2 C). The signal intensity on ADC sequence was mildly reduced ( Fig. 2 D). No obvious enhancement was found on Gd-DTPA enhanced images ( Fig. 2 E). MR spectroscopy was carried out as well. Compared to the ipsilateral normally appearing area ( Fig. 2 F), there was a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) with decreased NAA spectrum and reduced NAA/Cr ratio ( Fig. 2 G). The change of the spectrum reflected the severity of metabolic disorders, suggesting the local accumulation of lactic acid and disturbance of hypoxic processes. Fig. 1 CT scan showed lesions of hypodense in the left temporal and parietal lobe (Panel A). Three-dimensional reconstruction of CT vessel images (Panel B). Fig. 1 Fig. 2 Brain MRI (3.0 T) revealed a hypointense lesion in the left temporal and parietal lobe on T1 (Panel A), and increased signal intensity in the same region on Flair (Panel A) image with a clearly restricted diffusion (Panel C). The signal intensity on ADC sequence is mildly reduced (Panel D). No obvious enhancement was found on Gd-DTPA enhanced images (Panel E). Compared to the ipsilateral normally appearing area (Panel F), MR spectroscopy presented a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) (Panel G). Panels f and g are the molecular findings of metabolites respectively. Fig. 2 An electroencephalogram was performed on day 7 and displayed bilateral slow wave activities. Brainstem auditory evoked potential (BAEP) showed the bilateral sensorineural hearing loss. All the clinical and radiological findings were suggestive of mitochondrial disease. The final diagnosis of MELAS syndrome was confirmed by genetic analysis. The patient's peripheral blood leukocytes was detected by restriction fragment length polymorphism (RFLP) and revealed a mitochondrial DNA (mtDNA) mutation at A3242G point ( Fig. 3 ). Fig. 3 Mitochondrial DNA (mtDNA) mutation at A3242G point. Fig. 3 Her medications included L-arginine, phenobarbital, co-enzyme Q and levothyroxine substitution therapy. The patient's condition continuously improved, and was discharged on day 23. Six months following discharge, this patient had no further seizure."}
{'Case report': "The patient was a 37-year-old female, of 157 cm in height and 45 kg in weight. She was transferred to our department from a local hospital due to psychiatric features (both agitated behavior and auditory hallucinations), alexia and apraxia that had begun 10 days ago, followed by disorientation and generalized tonic-clonic seizures. She had a long history of episodic migraine-like headaches and progressive bilateral hearing loss for 3 years. However, she did not take any medication. Her vital signs showed a normal body temperature of 36.8 °C, a hypotension of 90/56 mm Hg and pulse at 72 beats per minute. Because of the patient's psychiatric symptoms and application of sedative after seizure attacks, she could not cooperate with physical examination. The laboratory data showed high anion gap metabolic acidosis with elevated levels of lactate and pyruvate. Serum levels of thyroid-stimulating hormone (TSH) and free thyroxine (FT4) were decreased. Her TSH level was low at 0.26 mU/L (normal range 0.35–5.5 mU/L), and FT4 concentration was 7.56 pmol/L (normal range 10.2–31 pmol/L). Both serum free and total triiodothyronine (FT3 and TT3) were significantly lower than normal range. FT3 concentration was 2.28 pmol/L (normal range 3.5–6.5 pmol/L), and TT3 concentration was 0.6 nmol/L (normal range 1.2–3.4 nmol/L). Moreover, elevated titers of serum anti-thyroglobulin and anti-thyroid microsomal antibodies were detected. Cerebral spinal fluid (CSF) studies were normal for cell counts and biochemistry, and negative for culture. CSF IgG index was 0.48 (normal range ≤ 0.7), and oligoclonal bands (OB) was negative. Computed tomography (CT) scan showed lesions of hypodense in the left temporal and parietal lobe, with brainstem and cerebellar atrophy ( Fig. 1 A). No evidence of subarachnoid or intracerebral hemorrhage. Vascular imaging of the cervical and cerebral arteries by CT angiography excluded the possibility of cerebrovascular disease ( Fig. 1 B). However, CT images were not conclusive to differentiate between the infectious or metabolic lesion. On day 5, brain magnetic resonance imaging MRI (3.0 T) revealed a hypointensity lesion in the left temporal-parietal lobe on T1 weighted image ( Fig. 2 A), and increased signal intensity in the same region on FLAIR sequences ( Fig. 2 B) with a clearly restricted diffusion ( Fig. 2 C). The signal intensity on ADC sequence was mildly reduced ( Fig. 2 D). No obvious enhancement was found on Gd-DTPA enhanced images ( Fig. 2 E). MR spectroscopy was carried out as well. Compared to the ipsilateral normally appearing area ( Fig. 2 F), there was a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) with decreased NAA spectrum and reduced NAA/Cr ratio ( Fig. 2 G). The change of the spectrum reflected the severity of metabolic disorders, suggesting the local accumulation of lactic acid and disturbance of hypoxic processes. Fig. 1 CT scan showed lesions of hypodense in the left temporal and parietal lobe (Panel A). Three-dimensional reconstruction of CT vessel images (Panel B). Fig. 1 Fig. 2 Brain MRI (3.0 T) revealed a hypointense lesion in the left temporal and parietal lobe on T1 (Panel A), and increased signal intensity in the same region on Flair (Panel A) image with a clearly restricted diffusion (Panel C). The signal intensity on ADC sequence is mildly reduced (Panel D). No obvious enhancement was found on Gd-DTPA enhanced images (Panel E). Compared to the ipsilateral normally appearing area (Panel F), MR spectroscopy presented a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) (Panel G). Panels f and g are the molecular findings of metabolites respectively. Fig. 2 An electroencephalogram was performed on day 7 and displayed bilateral slow wave activities. Brainstem auditory evoked potential (BAEP) showed the bilateral sensorineural hearing loss. All the clinical and radiological findings were suggestive of mitochondrial disease. The final diagnosis of MELAS syndrome was confirmed by genetic analysis. The patient's peripheral blood leukocytes was detected by restriction fragment length polymorphism (RFLP) and revealed a mitochondrial DNA (mtDNA) mutation at A3242G point ( Fig. 3 ). Fig. 3 Mitochondrial DNA (mtDNA) mutation at A3242G point. Fig. 3 Her medications included L-arginine, phenobarbital, co-enzyme Q and levothyroxine substitution therapy. The patient's condition continuously improved, and was discharged on day 23. Six months following discharge, this patient had no further seizure."}
Pregnancy
Pregnancy
[]
test-302
5721577
{'Case report': "The patient was a 37-year-old female, of 157 cm in height and 45 kg in weight. She was transferred to our department from a local hospital due to psychiatric features (both agitated behavior and auditory hallucinations), alexia and apraxia that had begun 10 days ago, followed by disorientation and generalized tonic-clonic seizures. She had a long history of episodic migraine-like headaches and progressive bilateral hearing loss for 3 years. However, she did not take any medication. Her vital signs showed a normal body temperature of 36.8 °C, a hypotension of 90/56 mm Hg and pulse at 72 beats per minute. Because of the patient's psychiatric symptoms and application of sedative after seizure attacks, she could not cooperate with physical examination. The laboratory data showed high anion gap metabolic acidosis with elevated levels of lactate and pyruvate. Serum levels of thyroid-stimulating hormone (TSH) and free thyroxine (FT4) were decreased. Her TSH level was low at 0.26 mU/L (normal range 0.35–5.5 mU/L), and FT4 concentration was 7.56 pmol/L (normal range 10.2–31 pmol/L). Both serum free and total triiodothyronine (FT3 and TT3) were significantly lower than normal range. FT3 concentration was 2.28 pmol/L (normal range 3.5–6.5 pmol/L), and TT3 concentration was 0.6 nmol/L (normal range 1.2–3.4 nmol/L). Moreover, elevated titers of serum anti-thyroglobulin and anti-thyroid microsomal antibodies were detected. Cerebral spinal fluid (CSF) studies were normal for cell counts and biochemistry, and negative for culture. CSF IgG index was 0.48 (normal range ≤ 0.7), and oligoclonal bands (OB) was negative. Computed tomography (CT) scan showed lesions of hypodense in the left temporal and parietal lobe, with brainstem and cerebellar atrophy ( Fig. 1 A). No evidence of subarachnoid or intracerebral hemorrhage. Vascular imaging of the cervical and cerebral arteries by CT angiography excluded the possibility of cerebrovascular disease ( Fig. 1 B). However, CT images were not conclusive to differentiate between the infectious or metabolic lesion. On day 5, brain magnetic resonance imaging MRI (3.0 T) revealed a hypointensity lesion in the left temporal-parietal lobe on T1 weighted image ( Fig. 2 A), and increased signal intensity in the same region on FLAIR sequences ( Fig. 2 B) with a clearly restricted diffusion ( Fig. 2 C). The signal intensity on ADC sequence was mildly reduced ( Fig. 2 D). No obvious enhancement was found on Gd-DTPA enhanced images ( Fig. 2 E). MR spectroscopy was carried out as well. Compared to the ipsilateral normally appearing area ( Fig. 2 F), there was a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) with decreased NAA spectrum and reduced NAA/Cr ratio ( Fig. 2 G). The change of the spectrum reflected the severity of metabolic disorders, suggesting the local accumulation of lactic acid and disturbance of hypoxic processes. Fig. 1 CT scan showed lesions of hypodense in the left temporal and parietal lobe (Panel A). Three-dimensional reconstruction of CT vessel images (Panel B). Fig. 1 Fig. 2 Brain MRI (3.0 T) revealed a hypointense lesion in the left temporal and parietal lobe on T1 (Panel A), and increased signal intensity in the same region on Flair (Panel A) image with a clearly restricted diffusion (Panel C). The signal intensity on ADC sequence is mildly reduced (Panel D). No obvious enhancement was found on Gd-DTPA enhanced images (Panel E). Compared to the ipsilateral normally appearing area (Panel F), MR spectroscopy presented a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) (Panel G). Panels f and g are the molecular findings of metabolites respectively. Fig. 2 An electroencephalogram was performed on day 7 and displayed bilateral slow wave activities. Brainstem auditory evoked potential (BAEP) showed the bilateral sensorineural hearing loss. All the clinical and radiological findings were suggestive of mitochondrial disease. The final diagnosis of MELAS syndrome was confirmed by genetic analysis. The patient's peripheral blood leukocytes was detected by restriction fragment length polymorphism (RFLP) and revealed a mitochondrial DNA (mtDNA) mutation at A3242G point ( Fig. 3 ). Fig. 3 Mitochondrial DNA (mtDNA) mutation at A3242G point. Fig. 3 Her medications included L-arginine, phenobarbital, co-enzyme Q and levothyroxine substitution therapy. The patient's condition continuously improved, and was discharged on day 23. Six months following discharge, this patient had no further seizure."}
{'Case report': "The patient was a 37-year-old female, of 157 cm in height and 45 kg in weight. She was transferred to our department from a local hospital due to psychiatric features (both agitated behavior and auditory hallucinations), alexia and apraxia that had begun 10 days ago, followed by disorientation and generalized tonic-clonic seizures. She had a long history of episodic migraine-like headaches and progressive bilateral hearing loss for 3 years. However, she did not take any medication. Her vital signs showed a normal body temperature of 36.8 °C, a hypotension of 90/56 mm Hg and pulse at 72 beats per minute. Because of the patient's psychiatric symptoms and application of sedative after seizure attacks, she could not cooperate with physical examination. The laboratory data showed high anion gap metabolic acidosis with elevated levels of lactate and pyruvate. Serum levels of thyroid-stimulating hormone (TSH) and free thyroxine (FT4) were decreased. Her TSH level was low at 0.26 mU/L (normal range 0.35–5.5 mU/L), and FT4 concentration was 7.56 pmol/L (normal range 10.2–31 pmol/L). Both serum free and total triiodothyronine (FT3 and TT3) were significantly lower than normal range. FT3 concentration was 2.28 pmol/L (normal range 3.5–6.5 pmol/L), and TT3 concentration was 0.6 nmol/L (normal range 1.2–3.4 nmol/L). Moreover, elevated titers of serum anti-thyroglobulin and anti-thyroid microsomal antibodies were detected. Cerebral spinal fluid (CSF) studies were normal for cell counts and biochemistry, and negative for culture. CSF IgG index was 0.48 (normal range ≤ 0.7), and oligoclonal bands (OB) was negative. Computed tomography (CT) scan showed lesions of hypodense in the left temporal and parietal lobe, with brainstem and cerebellar atrophy ( Fig. 1 A). No evidence of subarachnoid or intracerebral hemorrhage. Vascular imaging of the cervical and cerebral arteries by CT angiography excluded the possibility of cerebrovascular disease ( Fig. 1 B). However, CT images were not conclusive to differentiate between the infectious or metabolic lesion. On day 5, brain magnetic resonance imaging MRI (3.0 T) revealed a hypointensity lesion in the left temporal-parietal lobe on T1 weighted image ( Fig. 2 A), and increased signal intensity in the same region on FLAIR sequences ( Fig. 2 B) with a clearly restricted diffusion ( Fig. 2 C). The signal intensity on ADC sequence was mildly reduced ( Fig. 2 D). No obvious enhancement was found on Gd-DTPA enhanced images ( Fig. 2 E). MR spectroscopy was carried out as well. Compared to the ipsilateral normally appearing area ( Fig. 2 F), there was a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) with decreased NAA spectrum and reduced NAA/Cr ratio ( Fig. 2 G). The change of the spectrum reflected the severity of metabolic disorders, suggesting the local accumulation of lactic acid and disturbance of hypoxic processes. Fig. 1 CT scan showed lesions of hypodense in the left temporal and parietal lobe (Panel A). Three-dimensional reconstruction of CT vessel images (Panel B). Fig. 1 Fig. 2 Brain MRI (3.0 T) revealed a hypointense lesion in the left temporal and parietal lobe on T1 (Panel A), and increased signal intensity in the same region on Flair (Panel A) image with a clearly restricted diffusion (Panel C). The signal intensity on ADC sequence is mildly reduced (Panel D). No obvious enhancement was found on Gd-DTPA enhanced images (Panel E). Compared to the ipsilateral normally appearing area (Panel F), MR spectroscopy presented a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) (Panel G). Panels f and g are the molecular findings of metabolites respectively. Fig. 2 An electroencephalogram was performed on day 7 and displayed bilateral slow wave activities. Brainstem auditory evoked potential (BAEP) showed the bilateral sensorineural hearing loss. All the clinical and radiological findings were suggestive of mitochondrial disease. The final diagnosis of MELAS syndrome was confirmed by genetic analysis. The patient's peripheral blood leukocytes was detected by restriction fragment length polymorphism (RFLP) and revealed a mitochondrial DNA (mtDNA) mutation at A3242G point ( Fig. 3 ). Fig. 3 Mitochondrial DNA (mtDNA) mutation at A3242G point. Fig. 3 Her medications included L-arginine, phenobarbital, co-enzyme Q and levothyroxine substitution therapy. The patient's condition continuously improved, and was discharged on day 23. Six months following discharge, this patient had no further seizure."}
Lymphatic-System
LYMPH
[]
test-303
5721577
{'Case report': "The patient was a 37-year-old female, of 157 cm in height and 45 kg in weight. She was transferred to our department from a local hospital due to psychiatric features (both agitated behavior and auditory hallucinations), alexia and apraxia that had begun 10 days ago, followed by disorientation and generalized tonic-clonic seizures. She had a long history of episodic migraine-like headaches and progressive bilateral hearing loss for 3 years. However, she did not take any medication. Her vital signs showed a normal body temperature of 36.8 °C, a hypotension of 90/56 mm Hg and pulse at 72 beats per minute. Because of the patient's psychiatric symptoms and application of sedative after seizure attacks, she could not cooperate with physical examination. The laboratory data showed high anion gap metabolic acidosis with elevated levels of lactate and pyruvate. Serum levels of thyroid-stimulating hormone (TSH) and free thyroxine (FT4) were decreased. Her TSH level was low at 0.26 mU/L (normal range 0.35–5.5 mU/L), and FT4 concentration was 7.56 pmol/L (normal range 10.2–31 pmol/L). Both serum free and total triiodothyronine (FT3 and TT3) were significantly lower than normal range. FT3 concentration was 2.28 pmol/L (normal range 3.5–6.5 pmol/L), and TT3 concentration was 0.6 nmol/L (normal range 1.2–3.4 nmol/L). Moreover, elevated titers of serum anti-thyroglobulin and anti-thyroid microsomal antibodies were detected. Cerebral spinal fluid (CSF) studies were normal for cell counts and biochemistry, and negative for culture. CSF IgG index was 0.48 (normal range ≤ 0.7), and oligoclonal bands (OB) was negative. Computed tomography (CT) scan showed lesions of hypodense in the left temporal and parietal lobe, with brainstem and cerebellar atrophy ( Fig. 1 A). No evidence of subarachnoid or intracerebral hemorrhage. Vascular imaging of the cervical and cerebral arteries by CT angiography excluded the possibility of cerebrovascular disease ( Fig. 1 B). However, CT images were not conclusive to differentiate between the infectious or metabolic lesion. On day 5, brain magnetic resonance imaging MRI (3.0 T) revealed a hypointensity lesion in the left temporal-parietal lobe on T1 weighted image ( Fig. 2 A), and increased signal intensity in the same region on FLAIR sequences ( Fig. 2 B) with a clearly restricted diffusion ( Fig. 2 C). The signal intensity on ADC sequence was mildly reduced ( Fig. 2 D). No obvious enhancement was found on Gd-DTPA enhanced images ( Fig. 2 E). MR spectroscopy was carried out as well. Compared to the ipsilateral normally appearing area ( Fig. 2 F), there was a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) with decreased NAA spectrum and reduced NAA/Cr ratio ( Fig. 2 G). The change of the spectrum reflected the severity of metabolic disorders, suggesting the local accumulation of lactic acid and disturbance of hypoxic processes. Fig. 1 CT scan showed lesions of hypodense in the left temporal and parietal lobe (Panel A). Three-dimensional reconstruction of CT vessel images (Panel B). Fig. 1 Fig. 2 Brain MRI (3.0 T) revealed a hypointense lesion in the left temporal and parietal lobe on T1 (Panel A), and increased signal intensity in the same region on Flair (Panel A) image with a clearly restricted diffusion (Panel C). The signal intensity on ADC sequence is mildly reduced (Panel D). No obvious enhancement was found on Gd-DTPA enhanced images (Panel E). Compared to the ipsilateral normally appearing area (Panel F), MR spectroscopy presented a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) (Panel G). Panels f and g are the molecular findings of metabolites respectively. Fig. 2 An electroencephalogram was performed on day 7 and displayed bilateral slow wave activities. Brainstem auditory evoked potential (BAEP) showed the bilateral sensorineural hearing loss. All the clinical and radiological findings were suggestive of mitochondrial disease. The final diagnosis of MELAS syndrome was confirmed by genetic analysis. The patient's peripheral blood leukocytes was detected by restriction fragment length polymorphism (RFLP) and revealed a mitochondrial DNA (mtDNA) mutation at A3242G point ( Fig. 3 ). Fig. 3 Mitochondrial DNA (mtDNA) mutation at A3242G point. Fig. 3 Her medications included L-arginine, phenobarbital, co-enzyme Q and levothyroxine substitution therapy. The patient's condition continuously improved, and was discharged on day 23. Six months following discharge, this patient had no further seizure."}
{'Case report': "The patient was a 37-year-old female, of 157 cm in height and 45 kg in weight. She was transferred to our department from a local hospital due to psychiatric features (both agitated behavior and auditory hallucinations), alexia and apraxia that had begun 10 days ago, followed by disorientation and generalized tonic-clonic seizures. She had a long history of episodic migraine-like headaches and progressive bilateral hearing loss for 3 years. However, she did not take any medication. Her vital signs showed a normal body temperature of 36.8 °C, a hypotension of 90/56 mm Hg and pulse at 72 beats per minute. Because of the patient's psychiatric symptoms and application of sedative after seizure attacks, she could not cooperate with physical examination. The laboratory data showed high anion gap metabolic acidosis with elevated levels of lactate and pyruvate. Serum levels of thyroid-stimulating hormone (TSH) and free thyroxine (FT4) were decreased. Her TSH level was low at 0.26 mU/L (normal range 0.35–5.5 mU/L), and FT4 concentration was 7.56 pmol/L (normal range 10.2–31 pmol/L). Both serum free and total triiodothyronine (FT3 and TT3) were significantly lower than normal range. FT3 concentration was 2.28 pmol/L (normal range 3.5–6.5 pmol/L), and TT3 concentration was 0.6 nmol/L (normal range 1.2–3.4 nmol/L). Moreover, elevated titers of serum anti-thyroglobulin and anti-thyroid microsomal antibodies were detected. Cerebral spinal fluid (CSF) studies were normal for cell counts and biochemistry, and negative for culture. CSF IgG index was 0.48 (normal range ≤ 0.7), and oligoclonal bands (OB) was negative. Computed tomography (CT) scan showed lesions of hypodense in the left temporal and parietal lobe, with brainstem and cerebellar atrophy ( Fig. 1 A). No evidence of subarachnoid or intracerebral hemorrhage. Vascular imaging of the cervical and cerebral arteries by CT angiography excluded the possibility of cerebrovascular disease ( Fig. 1 B). However, CT images were not conclusive to differentiate between the infectious or metabolic lesion. On day 5, brain magnetic resonance imaging MRI (3.0 T) revealed a hypointensity lesion in the left temporal-parietal lobe on T1 weighted image ( Fig. 2 A), and increased signal intensity in the same region on FLAIR sequences ( Fig. 2 B) with a clearly restricted diffusion ( Fig. 2 C). The signal intensity on ADC sequence was mildly reduced ( Fig. 2 D). No obvious enhancement was found on Gd-DTPA enhanced images ( Fig. 2 E). MR spectroscopy was carried out as well. Compared to the ipsilateral normally appearing area ( Fig. 2 F), there was a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) with decreased NAA spectrum and reduced NAA/Cr ratio ( Fig. 2 G). The change of the spectrum reflected the severity of metabolic disorders, suggesting the local accumulation of lactic acid and disturbance of hypoxic processes. Fig. 1 CT scan showed lesions of hypodense in the left temporal and parietal lobe (Panel A). Three-dimensional reconstruction of CT vessel images (Panel B). Fig. 1 Fig. 2 Brain MRI (3.0 T) revealed a hypointense lesion in the left temporal and parietal lobe on T1 (Panel A), and increased signal intensity in the same region on Flair (Panel A) image with a clearly restricted diffusion (Panel C). The signal intensity on ADC sequence is mildly reduced (Panel D). No obvious enhancement was found on Gd-DTPA enhanced images (Panel E). Compared to the ipsilateral normally appearing area (Panel F), MR spectroscopy presented a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) (Panel G). Panels f and g are the molecular findings of metabolites respectively. Fig. 2 An electroencephalogram was performed on day 7 and displayed bilateral slow wave activities. Brainstem auditory evoked potential (BAEP) showed the bilateral sensorineural hearing loss. All the clinical and radiological findings were suggestive of mitochondrial disease. The final diagnosis of MELAS syndrome was confirmed by genetic analysis. The patient's peripheral blood leukocytes was detected by restriction fragment length polymorphism (RFLP) and revealed a mitochondrial DNA (mtDNA) mutation at A3242G point ( Fig. 3 ). Fig. 3 Mitochondrial DNA (mtDNA) mutation at A3242G point. Fig. 3 Her medications included L-arginine, phenobarbital, co-enzyme Q and levothyroxine substitution therapy. The patient's condition continuously improved, and was discharged on day 23. Six months following discharge, this patient had no further seizure."}
Age-at-Presentation
Age (at case presentation)
[ "37 - year - old" ]
test-304
5721577
{'Case report': "The patient was a 37-year-old female, of 157 cm in height and 45 kg in weight. She was transferred to our department from a local hospital due to psychiatric features (both agitated behavior and auditory hallucinations), alexia and apraxia that had begun 10 days ago, followed by disorientation and generalized tonic-clonic seizures. She had a long history of episodic migraine-like headaches and progressive bilateral hearing loss for 3 years. However, she did not take any medication. Her vital signs showed a normal body temperature of 36.8 °C, a hypotension of 90/56 mm Hg and pulse at 72 beats per minute. Because of the patient's psychiatric symptoms and application of sedative after seizure attacks, she could not cooperate with physical examination. The laboratory data showed high anion gap metabolic acidosis with elevated levels of lactate and pyruvate. Serum levels of thyroid-stimulating hormone (TSH) and free thyroxine (FT4) were decreased. Her TSH level was low at 0.26 mU/L (normal range 0.35–5.5 mU/L), and FT4 concentration was 7.56 pmol/L (normal range 10.2–31 pmol/L). Both serum free and total triiodothyronine (FT3 and TT3) were significantly lower than normal range. FT3 concentration was 2.28 pmol/L (normal range 3.5–6.5 pmol/L), and TT3 concentration was 0.6 nmol/L (normal range 1.2–3.4 nmol/L). Moreover, elevated titers of serum anti-thyroglobulin and anti-thyroid microsomal antibodies were detected. Cerebral spinal fluid (CSF) studies were normal for cell counts and biochemistry, and negative for culture. CSF IgG index was 0.48 (normal range ≤ 0.7), and oligoclonal bands (OB) was negative. Computed tomography (CT) scan showed lesions of hypodense in the left temporal and parietal lobe, with brainstem and cerebellar atrophy ( Fig. 1 A). No evidence of subarachnoid or intracerebral hemorrhage. Vascular imaging of the cervical and cerebral arteries by CT angiography excluded the possibility of cerebrovascular disease ( Fig. 1 B). However, CT images were not conclusive to differentiate between the infectious or metabolic lesion. On day 5, brain magnetic resonance imaging MRI (3.0 T) revealed a hypointensity lesion in the left temporal-parietal lobe on T1 weighted image ( Fig. 2 A), and increased signal intensity in the same region on FLAIR sequences ( Fig. 2 B) with a clearly restricted diffusion ( Fig. 2 C). The signal intensity on ADC sequence was mildly reduced ( Fig. 2 D). No obvious enhancement was found on Gd-DTPA enhanced images ( Fig. 2 E). MR spectroscopy was carried out as well. Compared to the ipsilateral normally appearing area ( Fig. 2 F), there was a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) with decreased NAA spectrum and reduced NAA/Cr ratio ( Fig. 2 G). The change of the spectrum reflected the severity of metabolic disorders, suggesting the local accumulation of lactic acid and disturbance of hypoxic processes. Fig. 1 CT scan showed lesions of hypodense in the left temporal and parietal lobe (Panel A). Three-dimensional reconstruction of CT vessel images (Panel B). Fig. 1 Fig. 2 Brain MRI (3.0 T) revealed a hypointense lesion in the left temporal and parietal lobe on T1 (Panel A), and increased signal intensity in the same region on Flair (Panel A) image with a clearly restricted diffusion (Panel C). The signal intensity on ADC sequence is mildly reduced (Panel D). No obvious enhancement was found on Gd-DTPA enhanced images (Panel E). Compared to the ipsilateral normally appearing area (Panel F), MR spectroscopy presented a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) (Panel G). Panels f and g are the molecular findings of metabolites respectively. Fig. 2 An electroencephalogram was performed on day 7 and displayed bilateral slow wave activities. Brainstem auditory evoked potential (BAEP) showed the bilateral sensorineural hearing loss. All the clinical and radiological findings were suggestive of mitochondrial disease. The final diagnosis of MELAS syndrome was confirmed by genetic analysis. The patient's peripheral blood leukocytes was detected by restriction fragment length polymorphism (RFLP) and revealed a mitochondrial DNA (mtDNA) mutation at A3242G point ( Fig. 3 ). Fig. 3 Mitochondrial DNA (mtDNA) mutation at A3242G point. Fig. 3 Her medications included L-arginine, phenobarbital, co-enzyme Q and levothyroxine substitution therapy. The patient's condition continuously improved, and was discharged on day 23. Six months following discharge, this patient had no further seizure."}
{'Case report': "The patient was a 37-year-old female, of 157 cm in height and 45 kg in weight. She was transferred to our department from a local hospital due to psychiatric features (both agitated behavior and auditory hallucinations), alexia and apraxia that had begun 10 days ago, followed by disorientation and generalized tonic-clonic seizures. She had a long history of episodic migraine-like headaches and progressive bilateral hearing loss for 3 years. However, she did not take any medication. Her vital signs showed a normal body temperature of 36.8 °C, a hypotension of 90/56 mm Hg and pulse at 72 beats per minute. Because of the patient's psychiatric symptoms and application of sedative after seizure attacks, she could not cooperate with physical examination. The laboratory data showed high anion gap metabolic acidosis with elevated levels of lactate and pyruvate. Serum levels of thyroid-stimulating hormone (TSH) and free thyroxine (FT4) were decreased. Her TSH level was low at 0.26 mU/L (normal range 0.35–5.5 mU/L), and FT4 concentration was 7.56 pmol/L (normal range 10.2–31 pmol/L). Both serum free and total triiodothyronine (FT3 and TT3) were significantly lower than normal range. FT3 concentration was 2.28 pmol/L (normal range 3.5–6.5 pmol/L), and TT3 concentration was 0.6 nmol/L (normal range 1.2–3.4 nmol/L). Moreover, elevated titers of serum anti-thyroglobulin and anti-thyroid microsomal antibodies were detected. Cerebral spinal fluid (CSF) studies were normal for cell counts and biochemistry, and negative for culture. CSF IgG index was 0.48 (normal range ≤ 0.7), and oligoclonal bands (OB) was negative. Computed tomography (CT) scan showed lesions of hypodense in the left temporal and parietal lobe, with brainstem and cerebellar atrophy ( Fig. 1 A). No evidence of subarachnoid or intracerebral hemorrhage. Vascular imaging of the cervical and cerebral arteries by CT angiography excluded the possibility of cerebrovascular disease ( Fig. 1 B). However, CT images were not conclusive to differentiate between the infectious or metabolic lesion. On day 5, brain magnetic resonance imaging MRI (3.0 T) revealed a hypointensity lesion in the left temporal-parietal lobe on T1 weighted image ( Fig. 2 A), and increased signal intensity in the same region on FLAIR sequences ( Fig. 2 B) with a clearly restricted diffusion ( Fig. 2 C). The signal intensity on ADC sequence was mildly reduced ( Fig. 2 D). No obvious enhancement was found on Gd-DTPA enhanced images ( Fig. 2 E). MR spectroscopy was carried out as well. Compared to the ipsilateral normally appearing area ( Fig. 2 F), there was a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) with decreased NAA spectrum and reduced NAA/Cr ratio ( Fig. 2 G). The change of the spectrum reflected the severity of metabolic disorders, suggesting the local accumulation of lactic acid and disturbance of hypoxic processes. Fig. 1 CT scan showed lesions of hypodense in the left temporal and parietal lobe (Panel A). Three-dimensional reconstruction of CT vessel images (Panel B). Fig. 1 Fig. 2 Brain MRI (3.0 T) revealed a hypointense lesion in the left temporal and parietal lobe on T1 (Panel A), and increased signal intensity in the same region on Flair (Panel A) image with a clearly restricted diffusion (Panel C). The signal intensity on ADC sequence is mildly reduced (Panel D). No obvious enhancement was found on Gd-DTPA enhanced images (Panel E). Compared to the ipsilateral normally appearing area (Panel F), MR spectroscopy presented a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) (Panel G). Panels f and g are the molecular findings of metabolites respectively. Fig. 2 An electroencephalogram was performed on day 7 and displayed bilateral slow wave activities. Brainstem auditory evoked potential (BAEP) showed the bilateral sensorineural hearing loss. All the clinical and radiological findings were suggestive of mitochondrial disease. The final diagnosis of MELAS syndrome was confirmed by genetic analysis. The patient's peripheral blood leukocytes was detected by restriction fragment length polymorphism (RFLP) and revealed a mitochondrial DNA (mtDNA) mutation at A3242G point ( Fig. 3 ). Fig. 3 Mitochondrial DNA (mtDNA) mutation at A3242G point. Fig. 3 Her medications included L-arginine, phenobarbital, co-enzyme Q and levothyroxine substitution therapy. The patient's condition continuously improved, and was discharged on day 23. Six months following discharge, this patient had no further seizure."}
Age-of-Onset
Age (of onset)
[]
test-305
5721577
{'Case report': "The patient was a 37-year-old female, of 157 cm in height and 45 kg in weight. She was transferred to our department from a local hospital due to psychiatric features (both agitated behavior and auditory hallucinations), alexia and apraxia that had begun 10 days ago, followed by disorientation and generalized tonic-clonic seizures. She had a long history of episodic migraine-like headaches and progressive bilateral hearing loss for 3 years. However, she did not take any medication. Her vital signs showed a normal body temperature of 36.8 °C, a hypotension of 90/56 mm Hg and pulse at 72 beats per minute. Because of the patient's psychiatric symptoms and application of sedative after seizure attacks, she could not cooperate with physical examination. The laboratory data showed high anion gap metabolic acidosis with elevated levels of lactate and pyruvate. Serum levels of thyroid-stimulating hormone (TSH) and free thyroxine (FT4) were decreased. Her TSH level was low at 0.26 mU/L (normal range 0.35–5.5 mU/L), and FT4 concentration was 7.56 pmol/L (normal range 10.2–31 pmol/L). Both serum free and total triiodothyronine (FT3 and TT3) were significantly lower than normal range. FT3 concentration was 2.28 pmol/L (normal range 3.5–6.5 pmol/L), and TT3 concentration was 0.6 nmol/L (normal range 1.2–3.4 nmol/L). Moreover, elevated titers of serum anti-thyroglobulin and anti-thyroid microsomal antibodies were detected. Cerebral spinal fluid (CSF) studies were normal for cell counts and biochemistry, and negative for culture. CSF IgG index was 0.48 (normal range ≤ 0.7), and oligoclonal bands (OB) was negative. Computed tomography (CT) scan showed lesions of hypodense in the left temporal and parietal lobe, with brainstem and cerebellar atrophy ( Fig. 1 A). No evidence of subarachnoid or intracerebral hemorrhage. Vascular imaging of the cervical and cerebral arteries by CT angiography excluded the possibility of cerebrovascular disease ( Fig. 1 B). However, CT images were not conclusive to differentiate between the infectious or metabolic lesion. On day 5, brain magnetic resonance imaging MRI (3.0 T) revealed a hypointensity lesion in the left temporal-parietal lobe on T1 weighted image ( Fig. 2 A), and increased signal intensity in the same region on FLAIR sequences ( Fig. 2 B) with a clearly restricted diffusion ( Fig. 2 C). The signal intensity on ADC sequence was mildly reduced ( Fig. 2 D). No obvious enhancement was found on Gd-DTPA enhanced images ( Fig. 2 E). MR spectroscopy was carried out as well. Compared to the ipsilateral normally appearing area ( Fig. 2 F), there was a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) with decreased NAA spectrum and reduced NAA/Cr ratio ( Fig. 2 G). The change of the spectrum reflected the severity of metabolic disorders, suggesting the local accumulation of lactic acid and disturbance of hypoxic processes. Fig. 1 CT scan showed lesions of hypodense in the left temporal and parietal lobe (Panel A). Three-dimensional reconstruction of CT vessel images (Panel B). Fig. 1 Fig. 2 Brain MRI (3.0 T) revealed a hypointense lesion in the left temporal and parietal lobe on T1 (Panel A), and increased signal intensity in the same region on Flair (Panel A) image with a clearly restricted diffusion (Panel C). The signal intensity on ADC sequence is mildly reduced (Panel D). No obvious enhancement was found on Gd-DTPA enhanced images (Panel E). Compared to the ipsilateral normally appearing area (Panel F), MR spectroscopy presented a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) (Panel G). Panels f and g are the molecular findings of metabolites respectively. Fig. 2 An electroencephalogram was performed on day 7 and displayed bilateral slow wave activities. Brainstem auditory evoked potential (BAEP) showed the bilateral sensorineural hearing loss. All the clinical and radiological findings were suggestive of mitochondrial disease. The final diagnosis of MELAS syndrome was confirmed by genetic analysis. The patient's peripheral blood leukocytes was detected by restriction fragment length polymorphism (RFLP) and revealed a mitochondrial DNA (mtDNA) mutation at A3242G point ( Fig. 3 ). Fig. 3 Mitochondrial DNA (mtDNA) mutation at A3242G point. Fig. 3 Her medications included L-arginine, phenobarbital, co-enzyme Q and levothyroxine substitution therapy. The patient's condition continuously improved, and was discharged on day 23. Six months following discharge, this patient had no further seizure."}
{'Case report': "The patient was a 37-year-old female, of 157 cm in height and 45 kg in weight. She was transferred to our department from a local hospital due to psychiatric features (both agitated behavior and auditory hallucinations), alexia and apraxia that had begun 10 days ago, followed by disorientation and generalized tonic-clonic seizures. She had a long history of episodic migraine-like headaches and progressive bilateral hearing loss for 3 years. However, she did not take any medication. Her vital signs showed a normal body temperature of 36.8 °C, a hypotension of 90/56 mm Hg and pulse at 72 beats per minute. Because of the patient's psychiatric symptoms and application of sedative after seizure attacks, she could not cooperate with physical examination. The laboratory data showed high anion gap metabolic acidosis with elevated levels of lactate and pyruvate. Serum levels of thyroid-stimulating hormone (TSH) and free thyroxine (FT4) were decreased. Her TSH level was low at 0.26 mU/L (normal range 0.35–5.5 mU/L), and FT4 concentration was 7.56 pmol/L (normal range 10.2–31 pmol/L). Both serum free and total triiodothyronine (FT3 and TT3) were significantly lower than normal range. FT3 concentration was 2.28 pmol/L (normal range 3.5–6.5 pmol/L), and TT3 concentration was 0.6 nmol/L (normal range 1.2–3.4 nmol/L). Moreover, elevated titers of serum anti-thyroglobulin and anti-thyroid microsomal antibodies were detected. Cerebral spinal fluid (CSF) studies were normal for cell counts and biochemistry, and negative for culture. CSF IgG index was 0.48 (normal range ≤ 0.7), and oligoclonal bands (OB) was negative. Computed tomography (CT) scan showed lesions of hypodense in the left temporal and parietal lobe, with brainstem and cerebellar atrophy ( Fig. 1 A). No evidence of subarachnoid or intracerebral hemorrhage. Vascular imaging of the cervical and cerebral arteries by CT angiography excluded the possibility of cerebrovascular disease ( Fig. 1 B). However, CT images were not conclusive to differentiate between the infectious or metabolic lesion. On day 5, brain magnetic resonance imaging MRI (3.0 T) revealed a hypointensity lesion in the left temporal-parietal lobe on T1 weighted image ( Fig. 2 A), and increased signal intensity in the same region on FLAIR sequences ( Fig. 2 B) with a clearly restricted diffusion ( Fig. 2 C). The signal intensity on ADC sequence was mildly reduced ( Fig. 2 D). No obvious enhancement was found on Gd-DTPA enhanced images ( Fig. 2 E). MR spectroscopy was carried out as well. Compared to the ipsilateral normally appearing area ( Fig. 2 F), there was a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) with decreased NAA spectrum and reduced NAA/Cr ratio ( Fig. 2 G). The change of the spectrum reflected the severity of metabolic disorders, suggesting the local accumulation of lactic acid and disturbance of hypoxic processes. Fig. 1 CT scan showed lesions of hypodense in the left temporal and parietal lobe (Panel A). Three-dimensional reconstruction of CT vessel images (Panel B). Fig. 1 Fig. 2 Brain MRI (3.0 T) revealed a hypointense lesion in the left temporal and parietal lobe on T1 (Panel A), and increased signal intensity in the same region on Flair (Panel A) image with a clearly restricted diffusion (Panel C). The signal intensity on ADC sequence is mildly reduced (Panel D). No obvious enhancement was found on Gd-DTPA enhanced images (Panel E). Compared to the ipsilateral normally appearing area (Panel F), MR spectroscopy presented a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) (Panel G). Panels f and g are the molecular findings of metabolites respectively. Fig. 2 An electroencephalogram was performed on day 7 and displayed bilateral slow wave activities. Brainstem auditory evoked potential (BAEP) showed the bilateral sensorineural hearing loss. All the clinical and radiological findings were suggestive of mitochondrial disease. The final diagnosis of MELAS syndrome was confirmed by genetic analysis. The patient's peripheral blood leukocytes was detected by restriction fragment length polymorphism (RFLP) and revealed a mitochondrial DNA (mtDNA) mutation at A3242G point ( Fig. 3 ). Fig. 3 Mitochondrial DNA (mtDNA) mutation at A3242G point. Fig. 3 Her medications included L-arginine, phenobarbital, co-enzyme Q and levothyroxine substitution therapy. The patient's condition continuously improved, and was discharged on day 23. Six months following discharge, this patient had no further seizure."}
Confirmed-Diagnosis-IEM
Confirmed_Diagnosis(IEM)
[ "The final diagnosis of MELAS syndrome was confirmed by genetic analysis ." ]
test-306
5721577
{'Case report': "The patient was a 37-year-old female, of 157 cm in height and 45 kg in weight. She was transferred to our department from a local hospital due to psychiatric features (both agitated behavior and auditory hallucinations), alexia and apraxia that had begun 10 days ago, followed by disorientation and generalized tonic-clonic seizures. She had a long history of episodic migraine-like headaches and progressive bilateral hearing loss for 3 years. However, she did not take any medication. Her vital signs showed a normal body temperature of 36.8 °C, a hypotension of 90/56 mm Hg and pulse at 72 beats per minute. Because of the patient's psychiatric symptoms and application of sedative after seizure attacks, she could not cooperate with physical examination. The laboratory data showed high anion gap metabolic acidosis with elevated levels of lactate and pyruvate. Serum levels of thyroid-stimulating hormone (TSH) and free thyroxine (FT4) were decreased. Her TSH level was low at 0.26 mU/L (normal range 0.35–5.5 mU/L), and FT4 concentration was 7.56 pmol/L (normal range 10.2–31 pmol/L). Both serum free and total triiodothyronine (FT3 and TT3) were significantly lower than normal range. FT3 concentration was 2.28 pmol/L (normal range 3.5–6.5 pmol/L), and TT3 concentration was 0.6 nmol/L (normal range 1.2–3.4 nmol/L). Moreover, elevated titers of serum anti-thyroglobulin and anti-thyroid microsomal antibodies were detected. Cerebral spinal fluid (CSF) studies were normal for cell counts and biochemistry, and negative for culture. CSF IgG index was 0.48 (normal range ≤ 0.7), and oligoclonal bands (OB) was negative. Computed tomography (CT) scan showed lesions of hypodense in the left temporal and parietal lobe, with brainstem and cerebellar atrophy ( Fig. 1 A). No evidence of subarachnoid or intracerebral hemorrhage. Vascular imaging of the cervical and cerebral arteries by CT angiography excluded the possibility of cerebrovascular disease ( Fig. 1 B). However, CT images were not conclusive to differentiate between the infectious or metabolic lesion. On day 5, brain magnetic resonance imaging MRI (3.0 T) revealed a hypointensity lesion in the left temporal-parietal lobe on T1 weighted image ( Fig. 2 A), and increased signal intensity in the same region on FLAIR sequences ( Fig. 2 B) with a clearly restricted diffusion ( Fig. 2 C). The signal intensity on ADC sequence was mildly reduced ( Fig. 2 D). No obvious enhancement was found on Gd-DTPA enhanced images ( Fig. 2 E). MR spectroscopy was carried out as well. Compared to the ipsilateral normally appearing area ( Fig. 2 F), there was a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) with decreased NAA spectrum and reduced NAA/Cr ratio ( Fig. 2 G). The change of the spectrum reflected the severity of metabolic disorders, suggesting the local accumulation of lactic acid and disturbance of hypoxic processes. Fig. 1 CT scan showed lesions of hypodense in the left temporal and parietal lobe (Panel A). Three-dimensional reconstruction of CT vessel images (Panel B). Fig. 1 Fig. 2 Brain MRI (3.0 T) revealed a hypointense lesion in the left temporal and parietal lobe on T1 (Panel A), and increased signal intensity in the same region on Flair (Panel A) image with a clearly restricted diffusion (Panel C). The signal intensity on ADC sequence is mildly reduced (Panel D). No obvious enhancement was found on Gd-DTPA enhanced images (Panel E). Compared to the ipsilateral normally appearing area (Panel F), MR spectroscopy presented a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) (Panel G). Panels f and g are the molecular findings of metabolites respectively. Fig. 2 An electroencephalogram was performed on day 7 and displayed bilateral slow wave activities. Brainstem auditory evoked potential (BAEP) showed the bilateral sensorineural hearing loss. All the clinical and radiological findings were suggestive of mitochondrial disease. The final diagnosis of MELAS syndrome was confirmed by genetic analysis. The patient's peripheral blood leukocytes was detected by restriction fragment length polymorphism (RFLP) and revealed a mitochondrial DNA (mtDNA) mutation at A3242G point ( Fig. 3 ). Fig. 3 Mitochondrial DNA (mtDNA) mutation at A3242G point. Fig. 3 Her medications included L-arginine, phenobarbital, co-enzyme Q and levothyroxine substitution therapy. The patient's condition continuously improved, and was discharged on day 23. Six months following discharge, this patient had no further seizure."}
{'Case report': "The patient was a 37-year-old female, of 157 cm in height and 45 kg in weight. She was transferred to our department from a local hospital due to psychiatric features (both agitated behavior and auditory hallucinations), alexia and apraxia that had begun 10 days ago, followed by disorientation and generalized tonic-clonic seizures. She had a long history of episodic migraine-like headaches and progressive bilateral hearing loss for 3 years. However, she did not take any medication. Her vital signs showed a normal body temperature of 36.8 °C, a hypotension of 90/56 mm Hg and pulse at 72 beats per minute. Because of the patient's psychiatric symptoms and application of sedative after seizure attacks, she could not cooperate with physical examination. The laboratory data showed high anion gap metabolic acidosis with elevated levels of lactate and pyruvate. Serum levels of thyroid-stimulating hormone (TSH) and free thyroxine (FT4) were decreased. Her TSH level was low at 0.26 mU/L (normal range 0.35–5.5 mU/L), and FT4 concentration was 7.56 pmol/L (normal range 10.2–31 pmol/L). Both serum free and total triiodothyronine (FT3 and TT3) were significantly lower than normal range. FT3 concentration was 2.28 pmol/L (normal range 3.5–6.5 pmol/L), and TT3 concentration was 0.6 nmol/L (normal range 1.2–3.4 nmol/L). Moreover, elevated titers of serum anti-thyroglobulin and anti-thyroid microsomal antibodies were detected. Cerebral spinal fluid (CSF) studies were normal for cell counts and biochemistry, and negative for culture. CSF IgG index was 0.48 (normal range ≤ 0.7), and oligoclonal bands (OB) was negative. Computed tomography (CT) scan showed lesions of hypodense in the left temporal and parietal lobe, with brainstem and cerebellar atrophy ( Fig. 1 A). No evidence of subarachnoid or intracerebral hemorrhage. Vascular imaging of the cervical and cerebral arteries by CT angiography excluded the possibility of cerebrovascular disease ( Fig. 1 B). However, CT images were not conclusive to differentiate between the infectious or metabolic lesion. On day 5, brain magnetic resonance imaging MRI (3.0 T) revealed a hypointensity lesion in the left temporal-parietal lobe on T1 weighted image ( Fig. 2 A), and increased signal intensity in the same region on FLAIR sequences ( Fig. 2 B) with a clearly restricted diffusion ( Fig. 2 C). The signal intensity on ADC sequence was mildly reduced ( Fig. 2 D). No obvious enhancement was found on Gd-DTPA enhanced images ( Fig. 2 E). MR spectroscopy was carried out as well. Compared to the ipsilateral normally appearing area ( Fig. 2 F), there was a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) with decreased NAA spectrum and reduced NAA/Cr ratio ( Fig. 2 G). The change of the spectrum reflected the severity of metabolic disorders, suggesting the local accumulation of lactic acid and disturbance of hypoxic processes. Fig. 1 CT scan showed lesions of hypodense in the left temporal and parietal lobe (Panel A). Three-dimensional reconstruction of CT vessel images (Panel B). Fig. 1 Fig. 2 Brain MRI (3.0 T) revealed a hypointense lesion in the left temporal and parietal lobe on T1 (Panel A), and increased signal intensity in the same region on Flair (Panel A) image with a clearly restricted diffusion (Panel C). The signal intensity on ADC sequence is mildly reduced (Panel D). No obvious enhancement was found on Gd-DTPA enhanced images (Panel E). Compared to the ipsilateral normally appearing area (Panel F), MR spectroscopy presented a significantly elevated lactate peak at 1.3 ppm in region of interest (ROI) (Panel G). Panels f and g are the molecular findings of metabolites respectively. Fig. 2 An electroencephalogram was performed on day 7 and displayed bilateral slow wave activities. Brainstem auditory evoked potential (BAEP) showed the bilateral sensorineural hearing loss. All the clinical and radiological findings were suggestive of mitochondrial disease. The final diagnosis of MELAS syndrome was confirmed by genetic analysis. The patient's peripheral blood leukocytes was detected by restriction fragment length polymorphism (RFLP) and revealed a mitochondrial DNA (mtDNA) mutation at A3242G point ( Fig. 3 ). Fig. 3 Mitochondrial DNA (mtDNA) mutation at A3242G point. Fig. 3 Her medications included L-arginine, phenobarbital, co-enzyme Q and levothyroxine substitution therapy. The patient's condition continuously improved, and was discharged on day 23. Six months following discharge, this patient had no further seizure."}
IEM-Treatment
IEM_Treatment
[ "L - arginine , phenobarbital , co - enzyme Q and levothyroxine substitution therapy ." ]
test-307
5415296
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
Vitals-and-Hematology
Vitals_Hema
[ "A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits" ]
test-308
5415296
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
Gastrointestinal-System
GI
[]
test-309
5415296
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
Patient-History
History
[ "A 7 - month - old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play - related problem. The owners ’ complaints were that the cat had never used the litter tray, and it did not know how to play", "After 3 months, the cat was referred to the neurology service again, in status epilepticus.", "After 2 years of treatment and a progressive worsening, the cat was euthanased." ]
test-310
5415296
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
Neurology
Neuro
[ "house soiling and a play - related problem.", "did not know how to play.", "the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘ tried to bite, catch and scratch the air ’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat ’s behavioural history was unremarkable", "moderate and hypermetric ataxic gait, and a bilateral lack of menace response", "based on the behaviours at home described by the owners ( the inability to find some toys, and the behaviour of ‘ scratching and biting the air ’ ), some degree of visual impairment was suspected but not confirmed", "The cat did not crash with objects either at home or at the consultation room", "brain magnetic resonance imaging ( MRI; 0.2 T ) and cerebrospinal fluid analysis showed no abnormalities.", "suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability.", "status epilepticus", "lack of bilateral menace response and cerebellar ataxic gait", "progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status ( probably seizure activity )", "seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting < 2 mins", "No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS." ]
test-311
5415296
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
Laboratory-and-Imaging
Lab_Image
[ "A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus / feline leukaemia virus test was negative.", "A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging ( MRI; 0.2 T ) and cerebrospinal fluid analysis showed no abnormalities.", "Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α - mannosidase, β - mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits.", "No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS.", "PrPsc inmunohistochemistry assay was performed, and the results were negative" ]
test-312
5415296
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
Cardiovascular-System
CVS
[]
test-313
5415296
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
Endocrinology
ENDO
[]
test-314
5415296
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
Genitourinary-System
GU
[]
test-315
5415296
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
Respiratory-System
RESP
[]
test-316
5415296
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
Musculoskeletal-System
MSK
[ "bilateral carpal valgus" ]
test-317
5415296
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
Eyes-Ears-Nose-Throat
EENT
[ "No ophthalmological abnormalities were detected", "based on the behaviours at home described by the owners ( the inability to find some toys, and the behaviour of ‘ scratching and biting the air ’ ), some degree of visual impairment was suspected but not confirmed", "visible suture line in the posterior capsule of both crystalline lenses were also detected" ]
test-318
5415296
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
Dermatology
DERM
[]
test-319
5415296
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
Pregnancy
Pregnancy
[]
test-320
5415296
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
Lymphatic-System
LYMPH
[]
test-321
5415296
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
Age-at-Presentation
Age (at case presentation)
[ "7 - month - old" ]
test-322
5415296
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
Age-of-Onset
Age (of onset)
[]
test-323
5415296
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
Confirmed-Diagnosis-IEM
Confirmed_Diagnosis(IEM)
[ "congenital spongiform polioencephalomyelopathy ( CSP ) was diagnosed postmortem" ]
test-324
5415296
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
{'Case Report': 'A 7-month-old, entire female, domestic shorthair cat was referred to our behavioural service owing to house soiling and a play-related problem. The owners’ complaints were that the cat had never used the litter tray, and it did not know how to play. The environment consisted of two young adult humans with no children. They lived in a flat of 85 m 2, with two terraces of 5 m 2 each. There were three separated litter boxes at home, all of which were non-covered with low sides. The owners had used clumping, non-clumping, silica-based and soil-based litter during the months between the adoption (when the cat was 4 months old) and the first visit. One of the latrines always had clumping substrate. There were three food and three water troughs, all of them far from the latrines. The impression of the owners was that the cat eliminated where it was at any given moment. It eliminated many more times in front of the owner (90%) than when it was alone (10%). The cat never tried to cover its faeces or urine after depositions. Occasionally, the owners punished the cat verbally and physically, but only when it eliminated in front of them. The substrates used by the cat were ceramic tiles, the sofa and beds. It always adopted an emptying-body posture. Spots were always located on horizontal surfaces. The owners used bleach-based products in order to clean the spots, and just water in the case of the sofa and beds. Regarding the play-related problem, the owners said that the cat did not understand the body language of other cats and commonly crashed into other cats or people. It also ‘tried to bite, catch and scratch the air’ when playing. It did not find balls or other toys when the owners threw them to the cat to play. The rest of the cat’s behavioural history was unremarkable. The differential diagnoses of the house-soiling problem included problems with the litter trays, including insufficient number, incorrect type, competition with other cats for the latrines, incorrect location, acquired aversion, or inappropriate substrate; a preference for another location or substrate; a problem with preference acquisition (ie, because of a cognitive impairment, or a sensory impairment or an unavailability of appropriate latrines or substrate during the first few weeks of life); and marking behaviours. Finally, a medical illness can contribute to all of these problems or to be the main cause. 1, 2 We could rule out most of these problems after interviewing the owners. Firstly, it was unlikely that there was a problem with the litter tray because the number, type and location were correct. The locations were correct because the animal eliminated near the litter tray if it was there. Many different substrates had been used. Secondly, it was not a problem of preference because the cat eliminated in different locations and surfaces. The age of the animal, the distribution of the spots and the body posture during elimination ruled out marking behaviours. Alterations in play behaviours described by the owners could have been due to a cognitive impairment, and/or a sensory impairment (ie, blindness). Play behaviours depend on learning capability and sensory systems. 3 Additionally, an enriched environment is necessary to learn and display play behaviours in a proper manner. 4 In that case, the social and instrumental environment was good. Regarding elimination, a problem of substrate preferences acquisition was diagnosed. A cognitive impairment, a medical condition or both could have been the cause of the problem during the elimination habits acquisition. Moreover, cognitive impairment and some medical conditions also could explain the play-related behavioural problems. During the consultation, the physical examination was unremarkable; however, the neurological examination revealed a moderate and hypermetric ataxic gait, and a bilateral lack of menace response. A complete ophthalmological examination was performed by the ophthalmological service in order to rule out ocular diseases. No ophthalmological abnormalities were detected. Additionally, based on the behaviours at home described by the owners (the inability to find some toys, and the behaviour of ‘scratching and biting the air’), some degree of visual impairment was suspected but not confirmed with the neurological examination. The cat did not crash with objects either at home or at the consultation room. The problem was located in the central nervous system (CNS); specifically, an intracranial and multifocal problem was diagnosed. A complete blood count and a complete biochemistry panel were performed, and all of the results were within normal limits. The feline immunodeficiency virus/feline leukaemia virus test was negative. A thorax radiograph, abdominal ultrasound, brain magnetic resonance imaging (MRI; 0.2 T) and cerebrospinal fluid analysis showed no abnormalities. Although some small lesions could be missed with low-field MRI, we had to assume the absence of lesions obtained in the work-up. Thus, a degenerative condition such as a lysosomal storage disease, organic aciduria or mitochondrial encephalopathy was suspected. Samples of blood and urine were sent to the University of Pennsylvania School of Veterinary Medicine for metabolic screen tests. Amino acids, organic acids, carbohydrates, nitroprusside, ketone and mucopolysaccharide concentrations were analysed, as was α-mannosidase, β-mannosidase, fucosidase and hexosaminidase A and B activity. All of these were within normal limits. No treatment was prescribed for the neurological problem. The owners were given advice on correcting the soiling problem in the house using reward-based training with a clicker. During the first week, the clicker was conditioned by a food reward, and the entire floor was covered with newspaper. Each time that the cat eliminated, it was rewarded with a clicker and food. Newspaper was removed progressively. Three months later, the cat used a small newspaper-covered area to eliminate. This partial improvement suggests that there was a learning impairment during the acquisition of habits but not a total lack of learning capability. After 3 months, the cat was referred to the neurology service again, in status epilepticus. Neurological findings, after the postictal phase, included a lack of bilateral menace response and cerebellar ataxic gait. A bilateral carpal valgus that had already been found in the first visit and a visible suture line in the posterior capsule of both crystalline lenses were also detected. The owners reported progressive gait deficits over the previous month and compulsive running episodes with a partially impaired mental status (probably seizure activity). A symptomatic treatment with diazepam (1 mg/kg intrarectally only if seizures) and phenobarbital (2 mg/kg PO q24h) was started, with a very poor response. After 15 days of treatment, levetiracetam was added (10 mg/kg PO q8h) owing to an increase in seizure activity, with an initially good response. However, seizures reappeared after 2 months with 1–2 episodes every 15 days each lasting <2 mins. After 2 years of treatment and a progressive worsening, the cat was euthanased. A complete necropsy was immediately performed. No gross lesions were found, and based on the CNS histological lesions shown in Figures 1 – 4, a diagnosis of spongiform polioencephalomyelopathy was made. The spongiform degeneration of the grey matter was extensively distributed in the whole CNS. In order to rule out prion aetiology, a PrPsc inmunohistochemistry assay was performed, and the results were negative. Thus, congenital spongiform polioencephalomyelopathy (CSP) was diagnosed postmortem. Spongy vacuolation seen by light microscopy in the neural tissue is defined as spongy degeneration, and may take the form of vacuoles within processes of the neuropil, vesiculation of myelin sheaths, or swelling of astrocyte or oligodendrocyte cytoplasm. 5 A congenital problem, retrovirus infection and prion disease have been suggested as possible aetiologies. 6 – 10 Congenital spongiform degeneration of the grey matter has been previously described in a few cases of cats; 6 – 8 however, the cause remains still unknown. Common clinical signs include gait alteration, seizures, blindness, bilateral cataracts, behavioural changes and cranial nerve alterations. These signs appear during very early stages of development (just after birth), with a progressive fatal outcome in a few days or months. Behavioural signs are poorly described in animal science literature. 6 – 8 Although the degeneration usually affects diffusely all the grey matter, the behavioural alterations and the evolution of the clinical signs depend on the affected area of the brain in each case. Nevertheless, the clinical signs do not always correlate with the degree of the histological lesions. A spongy degenerative problem of grey matter has also been described in humans, and occurs in isolated cases and in sibs. 11 – 13 In all of human cases reported, the problem appears early in infancy and the outcome is always fatal. All the affected children show learning disabilities (ie, retarded speech development) during the early periods of infancy. Additionally, they rapidly develop neurological signs, especially seizures. The clinical findings and neuropathological changes are very similar in humans and the present case. There are no studies regarding degeneration of grey matter and its effect on learning ability in animals. However, other neurodegenerative problems (ie, lysosomal storage diseases) and problems that lead to structural abnormalities of the forebrain (ie, hydrocephaly) may be correlated with learning disabilities in animals and humans. 14 – 17 The acquisition of the elimination habits occurs during the first weeks of a kitten’s life. Most kittens naturally seek out sand-like materials for elimination purposes. However, the preference for a substrate needs to be learnt during those first weeks of age. Learning disabilities and/or sensory impairments could modify the acquisition of these habits.'}
IEM-Treatment
IEM_Treatment
[]
test-325
5680934
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
Vitals-and-Hematology
Vitals_Hema
[]
test-326
5680934
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
Gastrointestinal-System
GI
[ "poor weight gain with swallowing dysfunction and significant reflux disease" ]
test-327
5680934
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
Patient-History
History
[ "This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia.", "He had poor weight gain with swallowing dysfunction and significant reflux disease", "He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age", "He developed central and obstructive sleep apneas at 3.5 years of age", "Parents were carriers of the mutation", "The parents ’ first child was diagnosed with steroid - resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks ’ gestation due to antenatal diagnosis of Ebstein ’s anomaly and multiple valvular abnormalities." ]
test-328
5680934
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
Neurology
Neuro
[ "He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia", "brachycephalic", "swallowing dysfunction", "started having orofacial and limb dyskinesias at 1.5 years", "refractory multifocal epilepsy at 3 years of age", "Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally", "magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal.", "has generalized dystonia, and is mainly chair - bound. He has intermittent eye contact and minimal vocalization" ]
test-329
5680934
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
Laboratory-and-Imaging
Lab_Image
[ "magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal.", "Whole - exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C > T ( p. Arg1044Cys ), c.3430C > T ( p. Arg1144Cys ), and c.4078G > A ( p. Ala1360Thr ) in the LRPPRC gene", "His muscle biopsy showed normal muscle architecture with no “ ragged - red ” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c - oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect", "His baseline lactates ranged from 1.5 to 3.6 mmol / L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke - like episodes" ]
test-330
5680934
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
Cardiovascular-System
CVS
[]
test-331
5680934
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
Endocrinology
ENDO
[]
test-332
5680934
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
Genitourinary-System
GU
[]
test-333
5680934
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
Respiratory-System
RESP
[ "central and obstructive sleep apneas at 3.5 years of age" ]
test-334
5680934
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
Musculoskeletal-System
MSK
[ "brachycephalic" ]
test-335
5680934
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
Eyes-Ears-Nose-Throat
EENT
[]
test-336
5680934
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
Dermatology
DERM
[ "hypopigmented hair" ]
test-337
5680934
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
Pregnancy
Pregnancy
[ "He was born at term, weighed 3100 g with good Apgar scores." ]
test-338
5680934
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
Lymphatic-System
LYMPH
[]
test-339
5680934
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
Age-at-Presentation
Age (at case presentation)
[ "5 years of age" ]
test-340
5680934
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
Age-of-Onset
Age (of onset)
[ "7 months of age" ]
test-341
5680934
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
Confirmed-Diagnosis-IEM
Confirmed_Diagnosis(IEM)
[]
test-342
5680934
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
{'Case Report': 'This boy was born to nonconsanguineous Chinese parents. He was born at term, weighed 3100 g with good Apgar scores. He had global developmental delay at 7 months of age with severe head lag and generalized hypotonia. He was brachycephalic with hypopigmented hair. He had poor weight gain with swallowing dysfunction and significant reflux disease requiring gastrostomy feeding. He started having orofacial and limb dyskinesias at 1.5 years of age and subsequently developed refractory multifocal epilepsy at 3 years of age requiring multiple antiepileptics and ketogenic diet to control his seizures. Electroencephalogram showed focal and diffuse slowing consistent with background encephalopathic state, with frequent multifocal epileptiform discharges bilaterally. He developed central and obstructive sleep apneas at 3.5 years of age and was initiated on night bilevel positive airway pressure. He was evaluated with magnetic resonance imaging brain scan and chromosomal microarray at 8 months of age, which was normal. Whole-exome sequencing and direct sequencing confirmed that he harbored compound heterozygous missense mutations c.3130C>T (p.Arg1044Cys), c.3430C>T (p.Arg1144Cys), and c.4078G>A (p.Ala1360Thr) in the LRPPRC gene mapped to chromosome 2p21-p16. Parents were carriers of the mutation. His muscle biopsy showed normal muscle architecture with no “ragged-red” fibers, necrotic fibers, or regenerating fibers seen. Cytochrome c-oxidase was positive in most of the fibers. There were no light microscopic or ultrastructural features to support mitochondrial myopathy. Respiratory chain enzymes in skeletal muscle were diagnostic for complex IV defect ( Table 1 ). He is currently 5 years of age, has generalized dystonia, and is mainly chair-bound. He has intermittent eye contact and minimal vocalization. His baseline lactates ranged from 1.5 to 3.6 mmol/L with mild intermittent metabolic acidosis. However, there were no episodes of acute ketosis, glycemic derangements, or any acute stroke-like episodes. The parents’ first child was diagnosed with steroid-resistant nephrotic syndrome at 18 months of age requiring tacrolimus treatment. Their second pregnancy was terminated at 22 weeks’ gestation due to antenatal diagnosis of Ebstein’s anomaly and multiple valvular abnormalities.'}
IEM-Treatment
IEM_Treatment
[]
test-343
6511931
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
Vitals-and-Hematology
Vitals_Hema
[ "' On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal" ]
test-344
6511931
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
Gastrointestinal-System
GI
[]
test-345
6511931
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
Patient-History
History
[ "A 52 - year - old female presented with a sudden onset of right - sided numbness and weakness that was accompanied by a left temporal cluster - like headache. No fever or prodromal infection was found at disease onset.", "Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word \" nothing \". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self - alleviation", "At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion / extension.", "The family and personal history was unremarkable.", "After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic - clonic seizure for 3 min occurred", "52 - year - old woman with recurrent stroke - like episodes" ]
test-346
6511931
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
Neurology
Neuro
[ "sudden onset of right - sided numbness and weakness that was accompanied by a left temporal cluster - like headache.", "Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word \" nothing \". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self - alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength", "At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion / extension.", "Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right - sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic - clonic seizure for 3 min occurred", "recurrent stroke - like episodes", "sudden onset of right - sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster - like headache and later developed epilepsy during hospitalization" ]
test-347
6511931
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
Laboratory-and-Imaging
Lab_Image
[ "MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion - weighted imaging and fluid - attenuated inversion recovery ( Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography ( Figure 1 ). Due to the stroke - like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity.", "Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow - up ( Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy ( MRS ), which revealed markedly elevated lactate ( Lac ) concentrations in the regions of interest in the left temporal lesion", "MRI scan showed a hyperintense signal abnormality in the right parietal lobe ( Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged - red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T > C mutation, with a heteroplasmy level of 69.6 %, in the mitochondrial complex I subunit gene MT - ND3. In contrast, this mutation was not found in her peripheral blood cells.", "Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions.", "Laboratory tests, including D - dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid ( CSF ) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges." ]
test-348
6511931
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
Cardiovascular-System
CVS
[]
test-349
6511931
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
Endocrinology
ENDO
[]
test-350
6511931
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
Genitourinary-System
GU
[]
test-351
6511931
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
Respiratory-System
RESP
[]
test-352
6511931
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
Musculoskeletal-System
MSK
[ "lack of symptoms of muscle weakness or pain", "no other signs suggestive of myopathy" ]
test-353
6511931
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
Eyes-Ears-Nose-Throat
EENT
[]
test-354
6511931
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
Dermatology
DERM
[]
test-355
6511931
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
Pregnancy
Pregnancy
[]
test-356
6511931
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
Lymphatic-System
LYMPH
[]
test-357
6511931
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
Age-at-Presentation
Age (at case presentation)
[ "52 - year - old" ]
test-358
6511931
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
Age-of-Onset
Age (of onset)
[]
test-359
6511931
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
Confirmed-Diagnosis-IEM
Confirmed_Diagnosis(IEM)
[ "m.10158T > C mutation in the MT - ND3 gene" ]
test-360
6511931
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
{'Chief complaints': 'A 52-year-old female presented with a sudden onset of right-sided numbness and weakness that was accompanied by a left temporal cluster-like headache. No fever or prodromal infection was found at disease onset.', 'Imaging examinations and history of present illness': 'MRI demonstrated a lamellar left parietal lobe lesion predominantly involving the cortex, with hyperintensity on both diffusion-weighted imaging and fluid-attenuated inversion recovery (Figure 1 ). The apparent diffusion coefficient map revealed a preserved, isointense signal. No abnormalities were found by susceptibility weighted imaging or magnetic resonance angiography and venography (Figure 1 ). Due to the stroke-like onset pattern and MRI features, further thrombophilia screening was performed and showed decreased protein S activity. A diagnosis of cortical venous thrombosis was first proposed. Mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) was also considered but temporarily excluded because of the incomplete manifestation and lack of genetic evidence. Anticoagulation therapy was initiated, and follow-up was performed to maintain the international normalized ratio (INR) within the target range. Two months later, the patient was readmitted for subacute cognitive impairment. She was unable to identify and communicate with family members; she also had difficulty understanding questions or instructions and instead responded by repeating the word "nothing". During hospitalization, a secondary generalized seizure occurred, initially with eyes gazing to the right and then convulsion developing, which lasted for approximately 10 s before self-alleviation. Neurological examination suggested transcortical sensory aphasia, with fully covered limb strength. Blood tests and CSF examination were normal; INR was 2.21. On repeated MRI, new lesions were identified in the left temporal lobe and were also detected 10 days later in the right temporal lobe on radiological follow-up (Figures 1 and 2 ). Although the MRI signal characteristics are consistent with the initial findings, the original lesion in the left parietal lobe had been alleviated, with cortical atrophy. We further conducted magnetic resonance spectroscopy (MRS), which revealed markedly elevated lactate (Lac) concentrations in the regions of interest in the left temporal lesion (Figure 1 ). Mitochondrial encephalopathy was diagnosed, and genetic testing using peripheral blood was performed. However, DNA testing for frequent MELAS and myoclonic epilepsy with ragged red fibers syndrome mutations were negative. Because of the lack of symptoms of muscle weakness or pain, the patient declined our suggestion of performing a muscle biopsy. Anticoagulation therapy was terminated, and levetiracetam (1000 mg/d) was administered. At 3 mo after her second admission, the patient was experiencing involuntary movement in her left limbs, with repetitive flexion/extension. An MRI scan showed a hyperintense signal abnormality in the right parietal lobe (Figure 2 ). Brachial biceps biopsy was performed. Histopathology revealed no abnormalities, and no necrotic or regenerating fibers were observed; ragged-red fibers and intense succinate dehydrogenase activity were not detected. Nonetheless, complete sequencing of mitochondrial DNA samples extracted from the biopsied muscle revealed a heteroplasmic m.10158T>C mutation, with a heteroplasmy level of 69.6%, in the mitochondrial complex I subunit gene MT-ND3 . In contrast, this mutation was not found in her peripheral blood cells.', 'Personal and family history': 'The family and personal history was unremarkable.', 'Physical examination upon admission': 'On physical examination, the height and weight of the patient were 154 cm and 56 kg, respectively. Vital signs were normal, as were heart, lung and abdominal exami-nations. Neurological examination showed intact mental status, with normal speech and comprehension. Mild 4/5 right-sided hemiparesis was present with normal tone in both the arm and leg, though no other focal neurological deficits were found. After admission, she complained of discomfort and tingling in the right leg, after which a generalized tonic-clonic seizure for 3 min occurred before it was stopped by a bolus of intravenous diazepam.', 'CASE SUMMARY': 'We report a 52-year-old woman with recurrent stroke-like episodes carrying the m.10158T>C mutation in the MT-ND3 gene, which is also responsible for fatal infant-onset Leigh syndrome. Despite the common mutation, the present case featured a distinct clinical and neuroimaging manifestation from Leigh syndrome. This patient presented with sudden onset of right-sided hemiparesis and hemilateral sensory disturbance accompanied by a left temporal cluster-like headache and later developed epilepsy during hospitalization, with no other signs suggestive of myopathy, lactate acidosis, or other systemic symptoms. Brain magnetic resonance imaging revealed variable lesions involving multiple cortical and subcortical regions. Furthermore, a negative genetic test obtained from peripheral blood delayed the diagnosis of mitochondrial disease, which was eventually established through second-generation DNA sequencing using biopsied muscle.', 'Laboratory examinations': 'Laboratory tests, including D-dimer, lactic acid, and serum autoantibody levels, as well as thyroid function and tumor markers indicated no apparent abnormalities. Glucose tolerance and lactic acid movement tolerance tests were normal. A lumbar puncture was performed, and her open intracranial pressure was 180 mm H 2 O. Cerebrospinal fluid (CSF) testing showed that cell counts and protein, glucose, chloride, monoclonal antibody, adenosine deaminase, and lactate dehydrogenas levels were within normal ranges.'}
IEM-Treatment
IEM_Treatment
[]
test-361
6971469
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
Vitals-and-Hematology
Vitals_Hema
[ "On admission to the ICU, the vital signs were as follows : body temperature, 39 ° C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths / min; and SpO2, 99 % ( FiO 2 40 % )" ]
test-362
6971469
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
Gastrointestinal-System
GI
[]
test-363
6971469
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
Patient-History
History
[ "A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus." ]
test-364
6971469
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
Neurology
Neuro
[ "mildly impaired consciousness and myoclonus in the extremities", "myoclonic movement in the extremities, mandible, and trunk", "Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging", "impaired consciousness and myoclonus persisted for 2 weeks", "brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex.", "Electroencephalography revealed high‐amplitude slow and spiked waves", "non‐convulsive status epilepticus", "impaired consciousness and myoclonus", "non‐convulsive status epilepticus" ]
test-365
6971469
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
Laboratory-and-Imaging
Lab_Image
[ "Genetic testing revealed a 3271 T > C transition in the MT‐TL1 gene", "A laboratory examination showed increased serum pyruvic acid ( 2.1 mg / dL ) and lactate ( 75 mg / dL )", "Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging", "brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex", "arterial blood gas revealed metabolic acidosis ( pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol / L, and lactate 99 mg / dL ). Creatinine kinase was elevated at 20,999 U / L.", "Laboratory findings showed metabolic acidosis ( lactic acid, 99 mg / dL; pyruvic acid, 0.92 mg / dL ), creatinine kinase 29,560 U / L, and myoglobin 9,845 ng / mL.", "metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU / L", "Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated." ]
test-366
6971469
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
Cardiovascular-System
CVS
[]
test-367
6971469
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
Endocrinology
ENDO
[]
test-368
6971469
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
Genitourinary-System
GU
[ "urine appeared brown", "PRIS" ]
test-369
6971469
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
Respiratory-System
RESP
[]
test-370
6971469
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
Musculoskeletal-System
MSK
[ "Muscle weakness was detected in the upper and lower limbs bilaterally", "urine appeared brown", "PRIS" ]
test-371
6971469
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
Eyes-Ears-Nose-Throat
EENT
[]
test-372
6971469
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
Dermatology
DERM
[]
test-373
6971469
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
Pregnancy
Pregnancy
[]
test-374
6971469
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
Lymphatic-System
LYMPH
[]
test-375
6971469
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
Age-at-Presentation
Age (at case presentation)
[ "24‐year‐old", "24‐year‐old" ]
test-376
6971469
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
Age-of-Onset
Age (of onset)
[]
test-377
6971469
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
Confirmed-Diagnosis-IEM
Confirmed_Diagnosis(IEM)
[ "diagnosed with MELAS at 21 years of age . Genetic testing revealed a 3271 T > C transition in the MT‐TL1 gene , clinically confirming the diagnosis of MELAS", "diagnosed with mitochondrial myopathy , encephalopathy , lactic acidosis , and stroke‐like episodes" ]
test-378
6971469
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
{'Case Report': 'A 24‐year‐old woman was admitted to our hospital with mildly impaired consciousness and myoclonus in the extremities. She had been diagnosed with MELAS at 21 years of age. Genetic testing revealed a 3271 T>C transition in the MT‐TL1 gene, clinically confirming the diagnosis of MELAS. After admission to hospital, neurological examination showed myoclonic movement in the extremities, mandible, and trunk. Muscle weakness was detected in the upper and lower limbs bilaterally. A laboratory examination showed increased serum pyruvic acid (2.1 mg/dL) and lactate (75 mg/dL). Brain magnetic resonance imaging showed bilateral high‐intensity lesions in the temporal and parietal lobes of the cortical and subcortical areas in T2 weighted imaging (Fig. 1 ). Antiepileptic drugs levetiracetam, perampanel, lacosamide, and clobazam were initiated. L‐arginine, coenzyme Q10, and L‐carnitine aimed at supporting mitochondrial energy production were given simultaneously. As the impaired consciousness and myoclonus persisted for 2 weeks, we undertook brain magnetic resonance imaging again, which revealed high‐intensity lesions in the right cortical temporal lobe, and arterial spin labeling showed increased blood flow in the bilateral frontal and temporal cortex. Electroencephalography revealed high‐amplitude slow and spiked waves. We diagnosed the patient with non‐convulsive status epilepticus. To control the seizures, propofol was given at a dose of 2.7 mg/kg/h for 12 h and then increased to 5.4 mg/kg/h, with midazolam added at a dose of 2 mg/h. On day 5, the urine appeared brown and arterial blood gas revealed metabolic acidosis (pH 7.261, pCO 2 33.0 mmHg, HCO 3 - 14.4 mmol/L, and lactate 99 mg/dL). Creatinine kinase was elevated at 20,999 U/L. The patient was diagnosed with PRIS. She was transferred to the intensive care unit (ICU) on day 19 of hospitalization. On admission to the ICU, the vital signs were as follows: body temperature, 39°C; blood pressure, 150/80 mmHg; heart rate, 150 b.p.m.; respiratory rate, 30 breaths/min; and SpO2, 99% (FiO 2 40%). Laboratory findings showed metabolic acidosis (lactic acid, 99 mg/dL; pyruvic acid, 0.92 mg/dL), creatinine kinase 29,560 U/L, and myoglobin 9,845 ng/mL. We treated her with interruption of propofol and continuous hemodiafiltration for metabolic acidosis. We continued coenzyme Q10, L‐carnitine, and L‐arginine for mitochondrial support for MELAS and antiepileptic drugs including levetiracetam, perampanel, lacosamide, and clobazam. After concurrent treatment of PRIS and MELAS, metabolic acidosis and creatinine kinase gradually decreased after reaching 59,000 IU/L on day 2 of ICU admission. Continuous hemodiafiltration was discontinued on day 11. Tracheostomy was carried out because i.v. midazolam treatment was prolonged to control the status epilepticus. Mechanical ventilation was discontinued on day 25 (Fig. 2 ). After rehabilitation, the patient was discharged on foot.', 'Case Presentation': 'A 24‐year‐old woman who had been diagnosed with mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke‐like episodes was admitted to our hospital with impaired consciousness and myoclonus. To control the non‐convulsive status epilepticus, propofol was administered. Arterial blood gas revealed metabolic acidosis, and creatinine kinase was elevated. The patient was diagnosed with PRIS. We treated her with interruption of propofol. She required mechanical ventilation for 25 days. After rehabilitation, she recovered and was discharged.'}
IEM-Treatment
IEM_Treatment
[ "L‐arginine , coenzyme Q10 , and L‐carnitine aimed at supporting mitochondrial energy production were given", "We continued coenzyme Q10 , L‐carnitine , and L‐arginine for mitochondrial support for MELAS" ]
test-379
6197309
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
Vitals-and-Hematology
Vitals_Hema
[ "normal vital signs and a body - mass index of 25 ( height : 1.65 m, weight : 68 kg )." ]
test-380
6197309
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
Gastrointestinal-System
GI
[]
test-381
6197309
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
Patient-History
History
[ "A 50 - year - old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score ( 24/25 ).", "past medical history was significant for a history of hypertension and hypercholesterolemia", "surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas", "family history was positive for lung and liver cancer in his mother" ]
test-382
6197309
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
Neurology
Neuro
[ "did not include cognitive impairment nor seizures", "neurological examination was normal except for mild muscle weakness at the end of the day" ]
test-383
6197309
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
Laboratory-and-Imaging
Lab_Image
[ "serum prostate specific antigen of 9.45 ng per milliliter on routine screening.", "urinalysis was negative for pyuria or microscopic hematuria", "common MERRF mitochondrial DNA mutation ( A8344 G ) in his blood", "prostate biopsy revealing grade group 2 adenocarcinoma ( Gleason score 3 + 4 = 7 ) at the left mid for 9 mm ( mm. ) and right base for 5.5 mm., and grade group 1 ( Gleason score 3 + 3 = 6 ) at the left base for 9 mm. Perineural invasion was present", "His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng / ml / cc.", "His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes.", "PSA was less than 0.1. ng / ml", "PSA remains less than 0.1 ng / ml. ' }" ]
test-384
6197309
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
Cardiovascular-System
CVS
[ "hypertension and hypercholesterolemia" ]
test-385
6197309
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
Endocrinology
ENDO
[]
test-386
6197309
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
Genitourinary-System
GU
[ "serum prostate specific antigen of 9.45 ng per milliliter on routine screening.", "mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35", "no erectile dysfunction with a normal International Index of Erectile Function score ( 24/25 ).", "normal external genitalia", "soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters", "His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng / ml / cc.", "high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement ( 9 mm. 5.5 mm., 9 mm. )", "His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes.", "PSA was less than 0.1. ng / ml.", "After 6 months, the patient could achieve intercourse with the aid of vardenafil on - demand with an International Index of Erectile function of 20/25", "At last follow - up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng / ml. '" ]
test-387
6197309
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
Respiratory-System
RESP
[]
test-388
6197309
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
Musculoskeletal-System
MSK
[ "exercise intolerance and mild muscle atrophy", "mild muscle weakness at the end of the day." ]
test-389
6197309
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
Eyes-Ears-Nose-Throat
EENT
[]
test-390
6197309
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
Dermatology
DERM
[ "symmetric lipomas in his cervical region", "multiple symmetric lipomatosis", "large, painless, symmetrical cervical lipomas", "cervical lipomas", "neck lipomas" ]
test-391
6197309
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
Pregnancy
Pregnancy
[]
test-392
6197309
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
Lymphatic-System
LYMPH
[]
test-393
6197309
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
Age-at-Presentation
Age (at case presentation)
[ "50 - year - old" ]
test-394
6197309
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
Age-of-Onset
Age (of onset)
[]
test-395
6197309
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
Confirmed-Diagnosis-IEM
Confirmed_Diagnosis(IEM)
[ "common MERRF mitochondrial DNA mutation ( A8344 G )" ]
test-396
6197309
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
{'Case presentation': 'A 50-year-old male with a history of MERRF syndrome was found to have serum prostate specific antigen of 9.45 ng per milliliter on routine screening. His urological history was significant for mild to moderate voiding dysfunction consisting of hesitancy and nocturia, with an International Prostate Symptom Score of 14/35. There was no erectile dysfunction with a normal International Index of Erectile Function score (24/25). His urinalysis was negative for pyuria or microscopic hematuria. The patient had been diagnosed with a sporadic form of MERRF as an adult after developing symmetric lipomas in his cervical region along with exercise intolerance and mild muscle atrophy. Despite having the common MERRF mitochondrial DNA mutation (A8344G) in his blood with multiple symmetric lipomatosis, his variant of MERFF did not include cognitive impairment nor seizures. 1 His past medical history was significant for a history of hypertension and hypercholesterolemia and his medications included levocarnitine, losartan, hydrochlorothiazide, and metoprolol. His surgical history included a tonsillectomy as a child and plastic surgery to remove neck lipomas. His family history was positive for lung and liver cancer in his mother. His physical examination showed normal vital signs and a body-mass index of 25 (height: 1.65 m, weight: 68 kg). Relevant findings included large, painless, symmetrical cervical lipomas ( Fig. 1 A and B), normal external genitalia, and a digital rectal examination revealing a soft prostate with good lateral definition bilaterally and an estimated volume of 30 cubic centimeters (cc.). A neurological examination was normal except for mild muscle weakness at the end of the day. Fig. 1 (A) Front and (B) lateral images of the patient with MERRF syndrome with characteristic cervical lipomas (with patient permission). Fig. 1 The patient underwent a 12-core ultrasound-guided transrectal prostate biopsy revealing grade group 2 adenocarcinoma (Gleason score 3 + 4 = 7) at the left mid for 9 mm (mm.) and right base for 5.5 mm., and grade group 1 (Gleason score 3 + 3 = 6) at the left base for 9 mm. Perineural invasion was present. His transrectal prostate ultrasound showed a prostate volume of 28.9 cc but no hypoechoic nodules. His calculated PSA density was 0.32 ng/ml/cc. After discussing all the treatment options, the patient chose a radical prostatectomy given his young age, high PSA density, moderate voiding dysfunction, and grade group 2 with significant involvement (9 mm. 5.5 mm., 9 mm.). Since proceeding with surgery was contingent upon neurology clearance, the patient was evaluated by his neurologist with a specialty in mitochondrial disorders. Given that his MERRF variant was only associated with mild myopathy, the patient was cleared for surgery with a willingness to accept a potentially higher risk of complications during intubation given his neck lipomas, as well as a theoretically higher risk of urinary incontinence or erectile dysfunction given the associated myopathy which characterizes the MERRF syndrome. The patient underwent a transperitoneal bilateral nerve-sparing robotic radical prostatectomy and pelvic lymphadenectomy without complications. There were no intubation-related complications; operative time was 150 minutes with an estimated blood loss 50 cc. The patient was discharged on postoperative day 1 without complications nor transfusions and his catheter was removed on postoperative day 7. His prostatectomy pathology was grade group 2, pathologic stage T2 with negative margins and lymph nodes. Based on the EPIC short form questionnaire administered four months after surgery the patient was free of pads and his PSA was less than 0.1. ng/ml. After 6 months, the patient could achieve intercourse with the aid of vardenafil on-demand with an International Index of Erectile function of 20/25. At last follow-up 52 months after surgery, the patient continues to be continent, has satisfactory sexual function using daily tadalafil, and his PSA remains less than 0.1 ng/ml.'}
IEM-Treatment
IEM_Treatment
[]
test-397
6302035
{'Case Presentation': "We present a case of cardiac paraganglioma with left coronary artery involvement. The patient was a 22-year-old woman with a clinical history of recurrent episodes of palpitations, diaphoresis, and headache. She was found to have high blood pressure and, because of her age, several studies were done looking for secondary hypertension. Results of transthoracic echocardiography and renal Doppler imaging were normal, and plasma and urine metanephrine levels were within normal limits. Ambulatory blood pressure monitoring found severe hypertension, and the patient started treatment with angiotensin receptor blockers. After 2 years, she continued to have the same symptoms, but at this time, a mesocardial systolic murmur with back irradiation was found on auscultation. New transthoracic echocardiography showed high velocities in the pulmonary artery and a mass with intermediate echogenicity located at the base of the heart in close relation to the aorta and the pulmonary artery, extending into the left atrioventricular groove ( Figure 1, Videos 1 and 2 ). Free metanephrines in plasma (noradrenaline 4,886.67 pg/mL) and urine (noradrenaline 1,426.9 μg/24 h) were high. Figure 1 Transthoracic echocardiography. Shown is a mass ( white arrow ) with intermediate echogenicity in relation to the great vessels. To obtain better characterization, cardiac magnetic resonance was performed and showed a middle mediastinal mass located in the left atrioventricular groove, extending posteriorly to the left atrial roof and almost surrounding the pulmonary artery anteriorly. The mass was closely associated with the aortic root, but the emergence and path of the left coronary artery and its branches were not adequately visualized ( Figure 2 ). Tissue characterization sequences showed a mass with a heterogeneous appearance on T2-weighted sequences and slightly hyperintense on T1-weighted and T2 with fat saturation sequences ( Figure 2 ). Important mass vascularization was demonstrated with the perfusion sequence, which showed rapid uptake of contrast shortly after its administration. We also found focal late gadolinium enhancement at the periphery of the mass, with no enhancement at its center, a finding that was considered highly suggestive of a paraganglioma ( Figure 3 ). Fluorine-18 fluorodeoxyglucose positron emission tomography was also performed, demonstrating a highly metabolic mass ( Figure 4 ). Figure 2 Cardiac magnetic resonance. (1) Coronal: isointense mass in relation to the left atrial roof. (2) Two-chamber longitudinal: isointense mass in the left atrioventricular groove. (3,4) Axial: mass in relation to the aortic root and the pulmonary artery. Images reproduced with permission from Clínica Shaio-Bogotá-Colombia. Figure 3 Cardiac magnetic resonance. Delayed enhancement. Focal deposit of gadolinium in the periphery of the mass. Images reproduced with permission from Clínica Shaio-Bogotá-Colombia. Figure 4 Positron emission tomography with 18F-fluorodeoxyglucose (10 mCi). Hypermetabolic mass in the middle mediastinum. Images reproduced with permission from Clínica del Country-Bogotá-Colombia. The case was discussed with the heart team and other departments, such as endocrinology and oncology, concluding that the patient had a clear indication for surgery, with α- and β-blockade, high sodium intake, and adequate hydration before the procedure. However, given that there were doubts regarding the emergence of the left coronary artery and its pathway, coronary angiotomography was performed. The mass was found to surround the left main coronary artery, the proximal segment of the left anterior descending coronary artery, the circumflex coronary artery, and two septal arteries, as well as the great cardiac vein ( Figure 5, Figure 6, Figure 7 ). At that time, and because of these findings, the intervention was considered to be a high-risk procedure. Isotopic radiation with 131 I meta-iodine benzyl guanidine (MIBG) was considered as a palliative intervention, but this approach was rejected because of the risk for hypertensive crises of unpredictable severity. Another approach that was considered was the use of octeotride, but this alternative was not accepted by the multidisciplinary group, because there was a risk for irradiation of adjacent tissue, including the coronary arteries, with an unknown benefit. The risk/benefit assessment of this therapy did not favor the patient either, and surgery was thought to be the only option. Coronary angiography was done looking for a main feeding artery that could be embolized preoperatively to reduce the tumor size and intraoperative blood loss. However, embolization could not be done, because there were many small feeding arteries coming from the left main coronary artery ( Figure 8 ). The patient underwent mass resection, requiring a complete transverse arteriotomy of the ascending aorta, pulmonary trunk, and section of the superior vena cava. The mass was exposed using caudal traction and was found to be closely associated with the posterior wall of the ascending aorta, the pulmonary artery, and the left atrial roof, without infiltration of these structures. Involvement of the left coronary artery trunk was documented, which required ligation and revascularization of the anterior descending coronary artery and the circumflex coronary artery. The mass could be completely resected ( Figure 9 ). Figure 5 Coronary computed tomography. Mass surrounding the left main coronary artery and the proximal segment of the anterior descending and the circumflex coronary arteries. Figure 6 Coronary computed tomography. Mass surrounding the pulmonary artery. Figure 7 Coronary computed tomography. Mass located in the left atrioventricular groove. Figure 8 Coronary arteriography. Mass irrigated by multiple small vessels arising from the anterior descending coronary artery. Figure 9 Surgical specimen. Resected mass. The patient progressed well and was able to be extubated and have her vasopressor support discontinued. Follow-up echocardiography showed that systolic function was moderately affected, with an ejection fraction of 35% and diffuse hypokinesia, which was more pronounced in the anterior descending coronary artery territory. Because of these findings, new coronary angiography was performed, ruling out saphenous bridge compromise ( Figure 10 ). Further follow-up echocardiography showed improvement in systolic function, with an ejection fraction of 55% and preserved contractility of all segments except the basal segment of the anterior septum, which continued to be akinetic. There was also moderate pericardial effusion without increased intrapericardial pressure ( Figure 11, Videos 3 and 4 ). Figure 10 Coronary angiography. Saphenous bridge patent to the anterior descending artery, without lesions. Figure 11 Transthoracic echocardiography. (1) Parasternal view in diastole: pericardial effusion. (2) Parasternal view in systole: akinesia of the basal anteroseptal segment. The pathology report of the surgical specimen described round and oval cells in an organoid pattern with fine chromatin nuclei and degenerative focal atypia ( Figure 12 ). These findings and the immunohistochemical studies were compatible with a paraganglioma. The genetic study showed a succinate dehydrogenase subunit B mutation. The patient progressed satisfactorily and was discharged. One month later, it was found that one of her father's cousins had a para-aortic pheochromocytoma, an incidental finding in the emergency department due to abdominal pain. Figure 12 Round and oval cells, arranged in an organoid pattern, with degenerative focal atypia."}
{'Case Presentation': "We present a case of cardiac paraganglioma with left coronary artery involvement. The patient was a 22-year-old woman with a clinical history of recurrent episodes of palpitations, diaphoresis, and headache. She was found to have high blood pressure and, because of her age, several studies were done looking for secondary hypertension. Results of transthoracic echocardiography and renal Doppler imaging were normal, and plasma and urine metanephrine levels were within normal limits. Ambulatory blood pressure monitoring found severe hypertension, and the patient started treatment with angiotensin receptor blockers. After 2 years, she continued to have the same symptoms, but at this time, a mesocardial systolic murmur with back irradiation was found on auscultation. New transthoracic echocardiography showed high velocities in the pulmonary artery and a mass with intermediate echogenicity located at the base of the heart in close relation to the aorta and the pulmonary artery, extending into the left atrioventricular groove ( Figure 1, Videos 1 and 2 ). Free metanephrines in plasma (noradrenaline 4,886.67 pg/mL) and urine (noradrenaline 1,426.9 μg/24 h) were high. Figure 1 Transthoracic echocardiography. Shown is a mass ( white arrow ) with intermediate echogenicity in relation to the great vessels. To obtain better characterization, cardiac magnetic resonance was performed and showed a middle mediastinal mass located in the left atrioventricular groove, extending posteriorly to the left atrial roof and almost surrounding the pulmonary artery anteriorly. The mass was closely associated with the aortic root, but the emergence and path of the left coronary artery and its branches were not adequately visualized ( Figure 2 ). Tissue characterization sequences showed a mass with a heterogeneous appearance on T2-weighted sequences and slightly hyperintense on T1-weighted and T2 with fat saturation sequences ( Figure 2 ). Important mass vascularization was demonstrated with the perfusion sequence, which showed rapid uptake of contrast shortly after its administration. We also found focal late gadolinium enhancement at the periphery of the mass, with no enhancement at its center, a finding that was considered highly suggestive of a paraganglioma ( Figure 3 ). Fluorine-18 fluorodeoxyglucose positron emission tomography was also performed, demonstrating a highly metabolic mass ( Figure 4 ). Figure 2 Cardiac magnetic resonance. (1) Coronal: isointense mass in relation to the left atrial roof. (2) Two-chamber longitudinal: isointense mass in the left atrioventricular groove. (3,4) Axial: mass in relation to the aortic root and the pulmonary artery. Images reproduced with permission from Clínica Shaio-Bogotá-Colombia. Figure 3 Cardiac magnetic resonance. Delayed enhancement. Focal deposit of gadolinium in the periphery of the mass. Images reproduced with permission from Clínica Shaio-Bogotá-Colombia. Figure 4 Positron emission tomography with 18F-fluorodeoxyglucose (10 mCi). Hypermetabolic mass in the middle mediastinum. Images reproduced with permission from Clínica del Country-Bogotá-Colombia. The case was discussed with the heart team and other departments, such as endocrinology and oncology, concluding that the patient had a clear indication for surgery, with α- and β-blockade, high sodium intake, and adequate hydration before the procedure. However, given that there were doubts regarding the emergence of the left coronary artery and its pathway, coronary angiotomography was performed. The mass was found to surround the left main coronary artery, the proximal segment of the left anterior descending coronary artery, the circumflex coronary artery, and two septal arteries, as well as the great cardiac vein ( Figure 5, Figure 6, Figure 7 ). At that time, and because of these findings, the intervention was considered to be a high-risk procedure. Isotopic radiation with 131 I meta-iodine benzyl guanidine (MIBG) was considered as a palliative intervention, but this approach was rejected because of the risk for hypertensive crises of unpredictable severity. Another approach that was considered was the use of octeotride, but this alternative was not accepted by the multidisciplinary group, because there was a risk for irradiation of adjacent tissue, including the coronary arteries, with an unknown benefit. The risk/benefit assessment of this therapy did not favor the patient either, and surgery was thought to be the only option. Coronary angiography was done looking for a main feeding artery that could be embolized preoperatively to reduce the tumor size and intraoperative blood loss. However, embolization could not be done, because there were many small feeding arteries coming from the left main coronary artery ( Figure 8 ). The patient underwent mass resection, requiring a complete transverse arteriotomy of the ascending aorta, pulmonary trunk, and section of the superior vena cava. The mass was exposed using caudal traction and was found to be closely associated with the posterior wall of the ascending aorta, the pulmonary artery, and the left atrial roof, without infiltration of these structures. Involvement of the left coronary artery trunk was documented, which required ligation and revascularization of the anterior descending coronary artery and the circumflex coronary artery. The mass could be completely resected ( Figure 9 ). Figure 5 Coronary computed tomography. Mass surrounding the left main coronary artery and the proximal segment of the anterior descending and the circumflex coronary arteries. Figure 6 Coronary computed tomography. Mass surrounding the pulmonary artery. Figure 7 Coronary computed tomography. Mass located in the left atrioventricular groove. Figure 8 Coronary arteriography. Mass irrigated by multiple small vessels arising from the anterior descending coronary artery. Figure 9 Surgical specimen. Resected mass. The patient progressed well and was able to be extubated and have her vasopressor support discontinued. Follow-up echocardiography showed that systolic function was moderately affected, with an ejection fraction of 35% and diffuse hypokinesia, which was more pronounced in the anterior descending coronary artery territory. Because of these findings, new coronary angiography was performed, ruling out saphenous bridge compromise ( Figure 10 ). Further follow-up echocardiography showed improvement in systolic function, with an ejection fraction of 55% and preserved contractility of all segments except the basal segment of the anterior septum, which continued to be akinetic. There was also moderate pericardial effusion without increased intrapericardial pressure ( Figure 11, Videos 3 and 4 ). Figure 10 Coronary angiography. Saphenous bridge patent to the anterior descending artery, without lesions. Figure 11 Transthoracic echocardiography. (1) Parasternal view in diastole: pericardial effusion. (2) Parasternal view in systole: akinesia of the basal anteroseptal segment. The pathology report of the surgical specimen described round and oval cells in an organoid pattern with fine chromatin nuclei and degenerative focal atypia ( Figure 12 ). These findings and the immunohistochemical studies were compatible with a paraganglioma. The genetic study showed a succinate dehydrogenase subunit B mutation. The patient progressed satisfactorily and was discharged. One month later, it was found that one of her father's cousins had a para-aortic pheochromocytoma, an incidental finding in the emergency department due to abdominal pain. Figure 12 Round and oval cells, arranged in an organoid pattern, with degenerative focal atypia."}
Vitals-and-Hematology
Vitals_Hema
[]
test-398
6302035
{'Case Presentation': "We present a case of cardiac paraganglioma with left coronary artery involvement. The patient was a 22-year-old woman with a clinical history of recurrent episodes of palpitations, diaphoresis, and headache. She was found to have high blood pressure and, because of her age, several studies were done looking for secondary hypertension. Results of transthoracic echocardiography and renal Doppler imaging were normal, and plasma and urine metanephrine levels were within normal limits. Ambulatory blood pressure monitoring found severe hypertension, and the patient started treatment with angiotensin receptor blockers. After 2 years, she continued to have the same symptoms, but at this time, a mesocardial systolic murmur with back irradiation was found on auscultation. New transthoracic echocardiography showed high velocities in the pulmonary artery and a mass with intermediate echogenicity located at the base of the heart in close relation to the aorta and the pulmonary artery, extending into the left atrioventricular groove ( Figure 1, Videos 1 and 2 ). Free metanephrines in plasma (noradrenaline 4,886.67 pg/mL) and urine (noradrenaline 1,426.9 μg/24 h) were high. Figure 1 Transthoracic echocardiography. Shown is a mass ( white arrow ) with intermediate echogenicity in relation to the great vessels. To obtain better characterization, cardiac magnetic resonance was performed and showed a middle mediastinal mass located in the left atrioventricular groove, extending posteriorly to the left atrial roof and almost surrounding the pulmonary artery anteriorly. The mass was closely associated with the aortic root, but the emergence and path of the left coronary artery and its branches were not adequately visualized ( Figure 2 ). Tissue characterization sequences showed a mass with a heterogeneous appearance on T2-weighted sequences and slightly hyperintense on T1-weighted and T2 with fat saturation sequences ( Figure 2 ). Important mass vascularization was demonstrated with the perfusion sequence, which showed rapid uptake of contrast shortly after its administration. We also found focal late gadolinium enhancement at the periphery of the mass, with no enhancement at its center, a finding that was considered highly suggestive of a paraganglioma ( Figure 3 ). Fluorine-18 fluorodeoxyglucose positron emission tomography was also performed, demonstrating a highly metabolic mass ( Figure 4 ). Figure 2 Cardiac magnetic resonance. (1) Coronal: isointense mass in relation to the left atrial roof. (2) Two-chamber longitudinal: isointense mass in the left atrioventricular groove. (3,4) Axial: mass in relation to the aortic root and the pulmonary artery. Images reproduced with permission from Clínica Shaio-Bogotá-Colombia. Figure 3 Cardiac magnetic resonance. Delayed enhancement. Focal deposit of gadolinium in the periphery of the mass. Images reproduced with permission from Clínica Shaio-Bogotá-Colombia. Figure 4 Positron emission tomography with 18F-fluorodeoxyglucose (10 mCi). Hypermetabolic mass in the middle mediastinum. Images reproduced with permission from Clínica del Country-Bogotá-Colombia. The case was discussed with the heart team and other departments, such as endocrinology and oncology, concluding that the patient had a clear indication for surgery, with α- and β-blockade, high sodium intake, and adequate hydration before the procedure. However, given that there were doubts regarding the emergence of the left coronary artery and its pathway, coronary angiotomography was performed. The mass was found to surround the left main coronary artery, the proximal segment of the left anterior descending coronary artery, the circumflex coronary artery, and two septal arteries, as well as the great cardiac vein ( Figure 5, Figure 6, Figure 7 ). At that time, and because of these findings, the intervention was considered to be a high-risk procedure. Isotopic radiation with 131 I meta-iodine benzyl guanidine (MIBG) was considered as a palliative intervention, but this approach was rejected because of the risk for hypertensive crises of unpredictable severity. Another approach that was considered was the use of octeotride, but this alternative was not accepted by the multidisciplinary group, because there was a risk for irradiation of adjacent tissue, including the coronary arteries, with an unknown benefit. The risk/benefit assessment of this therapy did not favor the patient either, and surgery was thought to be the only option. Coronary angiography was done looking for a main feeding artery that could be embolized preoperatively to reduce the tumor size and intraoperative blood loss. However, embolization could not be done, because there were many small feeding arteries coming from the left main coronary artery ( Figure 8 ). The patient underwent mass resection, requiring a complete transverse arteriotomy of the ascending aorta, pulmonary trunk, and section of the superior vena cava. The mass was exposed using caudal traction and was found to be closely associated with the posterior wall of the ascending aorta, the pulmonary artery, and the left atrial roof, without infiltration of these structures. Involvement of the left coronary artery trunk was documented, which required ligation and revascularization of the anterior descending coronary artery and the circumflex coronary artery. The mass could be completely resected ( Figure 9 ). Figure 5 Coronary computed tomography. Mass surrounding the left main coronary artery and the proximal segment of the anterior descending and the circumflex coronary arteries. Figure 6 Coronary computed tomography. Mass surrounding the pulmonary artery. Figure 7 Coronary computed tomography. Mass located in the left atrioventricular groove. Figure 8 Coronary arteriography. Mass irrigated by multiple small vessels arising from the anterior descending coronary artery. Figure 9 Surgical specimen. Resected mass. The patient progressed well and was able to be extubated and have her vasopressor support discontinued. Follow-up echocardiography showed that systolic function was moderately affected, with an ejection fraction of 35% and diffuse hypokinesia, which was more pronounced in the anterior descending coronary artery territory. Because of these findings, new coronary angiography was performed, ruling out saphenous bridge compromise ( Figure 10 ). Further follow-up echocardiography showed improvement in systolic function, with an ejection fraction of 55% and preserved contractility of all segments except the basal segment of the anterior septum, which continued to be akinetic. There was also moderate pericardial effusion without increased intrapericardial pressure ( Figure 11, Videos 3 and 4 ). Figure 10 Coronary angiography. Saphenous bridge patent to the anterior descending artery, without lesions. Figure 11 Transthoracic echocardiography. (1) Parasternal view in diastole: pericardial effusion. (2) Parasternal view in systole: akinesia of the basal anteroseptal segment. The pathology report of the surgical specimen described round and oval cells in an organoid pattern with fine chromatin nuclei and degenerative focal atypia ( Figure 12 ). These findings and the immunohistochemical studies were compatible with a paraganglioma. The genetic study showed a succinate dehydrogenase subunit B mutation. The patient progressed satisfactorily and was discharged. One month later, it was found that one of her father's cousins had a para-aortic pheochromocytoma, an incidental finding in the emergency department due to abdominal pain. Figure 12 Round and oval cells, arranged in an organoid pattern, with degenerative focal atypia."}
{'Case Presentation': "We present a case of cardiac paraganglioma with left coronary artery involvement. The patient was a 22-year-old woman with a clinical history of recurrent episodes of palpitations, diaphoresis, and headache. She was found to have high blood pressure and, because of her age, several studies were done looking for secondary hypertension. Results of transthoracic echocardiography and renal Doppler imaging were normal, and plasma and urine metanephrine levels were within normal limits. Ambulatory blood pressure monitoring found severe hypertension, and the patient started treatment with angiotensin receptor blockers. After 2 years, she continued to have the same symptoms, but at this time, a mesocardial systolic murmur with back irradiation was found on auscultation. New transthoracic echocardiography showed high velocities in the pulmonary artery and a mass with intermediate echogenicity located at the base of the heart in close relation to the aorta and the pulmonary artery, extending into the left atrioventricular groove ( Figure 1, Videos 1 and 2 ). Free metanephrines in plasma (noradrenaline 4,886.67 pg/mL) and urine (noradrenaline 1,426.9 μg/24 h) were high. Figure 1 Transthoracic echocardiography. Shown is a mass ( white arrow ) with intermediate echogenicity in relation to the great vessels. To obtain better characterization, cardiac magnetic resonance was performed and showed a middle mediastinal mass located in the left atrioventricular groove, extending posteriorly to the left atrial roof and almost surrounding the pulmonary artery anteriorly. The mass was closely associated with the aortic root, but the emergence and path of the left coronary artery and its branches were not adequately visualized ( Figure 2 ). Tissue characterization sequences showed a mass with a heterogeneous appearance on T2-weighted sequences and slightly hyperintense on T1-weighted and T2 with fat saturation sequences ( Figure 2 ). Important mass vascularization was demonstrated with the perfusion sequence, which showed rapid uptake of contrast shortly after its administration. We also found focal late gadolinium enhancement at the periphery of the mass, with no enhancement at its center, a finding that was considered highly suggestive of a paraganglioma ( Figure 3 ). Fluorine-18 fluorodeoxyglucose positron emission tomography was also performed, demonstrating a highly metabolic mass ( Figure 4 ). Figure 2 Cardiac magnetic resonance. (1) Coronal: isointense mass in relation to the left atrial roof. (2) Two-chamber longitudinal: isointense mass in the left atrioventricular groove. (3,4) Axial: mass in relation to the aortic root and the pulmonary artery. Images reproduced with permission from Clínica Shaio-Bogotá-Colombia. Figure 3 Cardiac magnetic resonance. Delayed enhancement. Focal deposit of gadolinium in the periphery of the mass. Images reproduced with permission from Clínica Shaio-Bogotá-Colombia. Figure 4 Positron emission tomography with 18F-fluorodeoxyglucose (10 mCi). Hypermetabolic mass in the middle mediastinum. Images reproduced with permission from Clínica del Country-Bogotá-Colombia. The case was discussed with the heart team and other departments, such as endocrinology and oncology, concluding that the patient had a clear indication for surgery, with α- and β-blockade, high sodium intake, and adequate hydration before the procedure. However, given that there were doubts regarding the emergence of the left coronary artery and its pathway, coronary angiotomography was performed. The mass was found to surround the left main coronary artery, the proximal segment of the left anterior descending coronary artery, the circumflex coronary artery, and two septal arteries, as well as the great cardiac vein ( Figure 5, Figure 6, Figure 7 ). At that time, and because of these findings, the intervention was considered to be a high-risk procedure. Isotopic radiation with 131 I meta-iodine benzyl guanidine (MIBG) was considered as a palliative intervention, but this approach was rejected because of the risk for hypertensive crises of unpredictable severity. Another approach that was considered was the use of octeotride, but this alternative was not accepted by the multidisciplinary group, because there was a risk for irradiation of adjacent tissue, including the coronary arteries, with an unknown benefit. The risk/benefit assessment of this therapy did not favor the patient either, and surgery was thought to be the only option. Coronary angiography was done looking for a main feeding artery that could be embolized preoperatively to reduce the tumor size and intraoperative blood loss. However, embolization could not be done, because there were many small feeding arteries coming from the left main coronary artery ( Figure 8 ). The patient underwent mass resection, requiring a complete transverse arteriotomy of the ascending aorta, pulmonary trunk, and section of the superior vena cava. The mass was exposed using caudal traction and was found to be closely associated with the posterior wall of the ascending aorta, the pulmonary artery, and the left atrial roof, without infiltration of these structures. Involvement of the left coronary artery trunk was documented, which required ligation and revascularization of the anterior descending coronary artery and the circumflex coronary artery. The mass could be completely resected ( Figure 9 ). Figure 5 Coronary computed tomography. Mass surrounding the left main coronary artery and the proximal segment of the anterior descending and the circumflex coronary arteries. Figure 6 Coronary computed tomography. Mass surrounding the pulmonary artery. Figure 7 Coronary computed tomography. Mass located in the left atrioventricular groove. Figure 8 Coronary arteriography. Mass irrigated by multiple small vessels arising from the anterior descending coronary artery. Figure 9 Surgical specimen. Resected mass. The patient progressed well and was able to be extubated and have her vasopressor support discontinued. Follow-up echocardiography showed that systolic function was moderately affected, with an ejection fraction of 35% and diffuse hypokinesia, which was more pronounced in the anterior descending coronary artery territory. Because of these findings, new coronary angiography was performed, ruling out saphenous bridge compromise ( Figure 10 ). Further follow-up echocardiography showed improvement in systolic function, with an ejection fraction of 55% and preserved contractility of all segments except the basal segment of the anterior septum, which continued to be akinetic. There was also moderate pericardial effusion without increased intrapericardial pressure ( Figure 11, Videos 3 and 4 ). Figure 10 Coronary angiography. Saphenous bridge patent to the anterior descending artery, without lesions. Figure 11 Transthoracic echocardiography. (1) Parasternal view in diastole: pericardial effusion. (2) Parasternal view in systole: akinesia of the basal anteroseptal segment. The pathology report of the surgical specimen described round and oval cells in an organoid pattern with fine chromatin nuclei and degenerative focal atypia ( Figure 12 ). These findings and the immunohistochemical studies were compatible with a paraganglioma. The genetic study showed a succinate dehydrogenase subunit B mutation. The patient progressed satisfactorily and was discharged. One month later, it was found that one of her father's cousins had a para-aortic pheochromocytoma, an incidental finding in the emergency department due to abdominal pain. Figure 12 Round and oval cells, arranged in an organoid pattern, with degenerative focal atypia."}
Gastrointestinal-System
GI
[]
test-399
6302035
{'Case Presentation': "We present a case of cardiac paraganglioma with left coronary artery involvement. The patient was a 22-year-old woman with a clinical history of recurrent episodes of palpitations, diaphoresis, and headache. She was found to have high blood pressure and, because of her age, several studies were done looking for secondary hypertension. Results of transthoracic echocardiography and renal Doppler imaging were normal, and plasma and urine metanephrine levels were within normal limits. Ambulatory blood pressure monitoring found severe hypertension, and the patient started treatment with angiotensin receptor blockers. After 2 years, she continued to have the same symptoms, but at this time, a mesocardial systolic murmur with back irradiation was found on auscultation. New transthoracic echocardiography showed high velocities in the pulmonary artery and a mass with intermediate echogenicity located at the base of the heart in close relation to the aorta and the pulmonary artery, extending into the left atrioventricular groove ( Figure 1, Videos 1 and 2 ). Free metanephrines in plasma (noradrenaline 4,886.67 pg/mL) and urine (noradrenaline 1,426.9 μg/24 h) were high. Figure 1 Transthoracic echocardiography. Shown is a mass ( white arrow ) with intermediate echogenicity in relation to the great vessels. To obtain better characterization, cardiac magnetic resonance was performed and showed a middle mediastinal mass located in the left atrioventricular groove, extending posteriorly to the left atrial roof and almost surrounding the pulmonary artery anteriorly. The mass was closely associated with the aortic root, but the emergence and path of the left coronary artery and its branches were not adequately visualized ( Figure 2 ). Tissue characterization sequences showed a mass with a heterogeneous appearance on T2-weighted sequences and slightly hyperintense on T1-weighted and T2 with fat saturation sequences ( Figure 2 ). Important mass vascularization was demonstrated with the perfusion sequence, which showed rapid uptake of contrast shortly after its administration. We also found focal late gadolinium enhancement at the periphery of the mass, with no enhancement at its center, a finding that was considered highly suggestive of a paraganglioma ( Figure 3 ). Fluorine-18 fluorodeoxyglucose positron emission tomography was also performed, demonstrating a highly metabolic mass ( Figure 4 ). Figure 2 Cardiac magnetic resonance. (1) Coronal: isointense mass in relation to the left atrial roof. (2) Two-chamber longitudinal: isointense mass in the left atrioventricular groove. (3,4) Axial: mass in relation to the aortic root and the pulmonary artery. Images reproduced with permission from Clínica Shaio-Bogotá-Colombia. Figure 3 Cardiac magnetic resonance. Delayed enhancement. Focal deposit of gadolinium in the periphery of the mass. Images reproduced with permission from Clínica Shaio-Bogotá-Colombia. Figure 4 Positron emission tomography with 18F-fluorodeoxyglucose (10 mCi). Hypermetabolic mass in the middle mediastinum. Images reproduced with permission from Clínica del Country-Bogotá-Colombia. The case was discussed with the heart team and other departments, such as endocrinology and oncology, concluding that the patient had a clear indication for surgery, with α- and β-blockade, high sodium intake, and adequate hydration before the procedure. However, given that there were doubts regarding the emergence of the left coronary artery and its pathway, coronary angiotomography was performed. The mass was found to surround the left main coronary artery, the proximal segment of the left anterior descending coronary artery, the circumflex coronary artery, and two septal arteries, as well as the great cardiac vein ( Figure 5, Figure 6, Figure 7 ). At that time, and because of these findings, the intervention was considered to be a high-risk procedure. Isotopic radiation with 131 I meta-iodine benzyl guanidine (MIBG) was considered as a palliative intervention, but this approach was rejected because of the risk for hypertensive crises of unpredictable severity. Another approach that was considered was the use of octeotride, but this alternative was not accepted by the multidisciplinary group, because there was a risk for irradiation of adjacent tissue, including the coronary arteries, with an unknown benefit. The risk/benefit assessment of this therapy did not favor the patient either, and surgery was thought to be the only option. Coronary angiography was done looking for a main feeding artery that could be embolized preoperatively to reduce the tumor size and intraoperative blood loss. However, embolization could not be done, because there were many small feeding arteries coming from the left main coronary artery ( Figure 8 ). The patient underwent mass resection, requiring a complete transverse arteriotomy of the ascending aorta, pulmonary trunk, and section of the superior vena cava. The mass was exposed using caudal traction and was found to be closely associated with the posterior wall of the ascending aorta, the pulmonary artery, and the left atrial roof, without infiltration of these structures. Involvement of the left coronary artery trunk was documented, which required ligation and revascularization of the anterior descending coronary artery and the circumflex coronary artery. The mass could be completely resected ( Figure 9 ). Figure 5 Coronary computed tomography. Mass surrounding the left main coronary artery and the proximal segment of the anterior descending and the circumflex coronary arteries. Figure 6 Coronary computed tomography. Mass surrounding the pulmonary artery. Figure 7 Coronary computed tomography. Mass located in the left atrioventricular groove. Figure 8 Coronary arteriography. Mass irrigated by multiple small vessels arising from the anterior descending coronary artery. Figure 9 Surgical specimen. Resected mass. The patient progressed well and was able to be extubated and have her vasopressor support discontinued. Follow-up echocardiography showed that systolic function was moderately affected, with an ejection fraction of 35% and diffuse hypokinesia, which was more pronounced in the anterior descending coronary artery territory. Because of these findings, new coronary angiography was performed, ruling out saphenous bridge compromise ( Figure 10 ). Further follow-up echocardiography showed improvement in systolic function, with an ejection fraction of 55% and preserved contractility of all segments except the basal segment of the anterior septum, which continued to be akinetic. There was also moderate pericardial effusion without increased intrapericardial pressure ( Figure 11, Videos 3 and 4 ). Figure 10 Coronary angiography. Saphenous bridge patent to the anterior descending artery, without lesions. Figure 11 Transthoracic echocardiography. (1) Parasternal view in diastole: pericardial effusion. (2) Parasternal view in systole: akinesia of the basal anteroseptal segment. The pathology report of the surgical specimen described round and oval cells in an organoid pattern with fine chromatin nuclei and degenerative focal atypia ( Figure 12 ). These findings and the immunohistochemical studies were compatible with a paraganglioma. The genetic study showed a succinate dehydrogenase subunit B mutation. The patient progressed satisfactorily and was discharged. One month later, it was found that one of her father's cousins had a para-aortic pheochromocytoma, an incidental finding in the emergency department due to abdominal pain. Figure 12 Round and oval cells, arranged in an organoid pattern, with degenerative focal atypia."}
{'Case Presentation': "We present a case of cardiac paraganglioma with left coronary artery involvement. The patient was a 22-year-old woman with a clinical history of recurrent episodes of palpitations, diaphoresis, and headache. She was found to have high blood pressure and, because of her age, several studies were done looking for secondary hypertension. Results of transthoracic echocardiography and renal Doppler imaging were normal, and plasma and urine metanephrine levels were within normal limits. Ambulatory blood pressure monitoring found severe hypertension, and the patient started treatment with angiotensin receptor blockers. After 2 years, she continued to have the same symptoms, but at this time, a mesocardial systolic murmur with back irradiation was found on auscultation. New transthoracic echocardiography showed high velocities in the pulmonary artery and a mass with intermediate echogenicity located at the base of the heart in close relation to the aorta and the pulmonary artery, extending into the left atrioventricular groove ( Figure 1, Videos 1 and 2 ). Free metanephrines in plasma (noradrenaline 4,886.67 pg/mL) and urine (noradrenaline 1,426.9 μg/24 h) were high. Figure 1 Transthoracic echocardiography. Shown is a mass ( white arrow ) with intermediate echogenicity in relation to the great vessels. To obtain better characterization, cardiac magnetic resonance was performed and showed a middle mediastinal mass located in the left atrioventricular groove, extending posteriorly to the left atrial roof and almost surrounding the pulmonary artery anteriorly. The mass was closely associated with the aortic root, but the emergence and path of the left coronary artery and its branches were not adequately visualized ( Figure 2 ). Tissue characterization sequences showed a mass with a heterogeneous appearance on T2-weighted sequences and slightly hyperintense on T1-weighted and T2 with fat saturation sequences ( Figure 2 ). Important mass vascularization was demonstrated with the perfusion sequence, which showed rapid uptake of contrast shortly after its administration. We also found focal late gadolinium enhancement at the periphery of the mass, with no enhancement at its center, a finding that was considered highly suggestive of a paraganglioma ( Figure 3 ). Fluorine-18 fluorodeoxyglucose positron emission tomography was also performed, demonstrating a highly metabolic mass ( Figure 4 ). Figure 2 Cardiac magnetic resonance. (1) Coronal: isointense mass in relation to the left atrial roof. (2) Two-chamber longitudinal: isointense mass in the left atrioventricular groove. (3,4) Axial: mass in relation to the aortic root and the pulmonary artery. Images reproduced with permission from Clínica Shaio-Bogotá-Colombia. Figure 3 Cardiac magnetic resonance. Delayed enhancement. Focal deposit of gadolinium in the periphery of the mass. Images reproduced with permission from Clínica Shaio-Bogotá-Colombia. Figure 4 Positron emission tomography with 18F-fluorodeoxyglucose (10 mCi). Hypermetabolic mass in the middle mediastinum. Images reproduced with permission from Clínica del Country-Bogotá-Colombia. The case was discussed with the heart team and other departments, such as endocrinology and oncology, concluding that the patient had a clear indication for surgery, with α- and β-blockade, high sodium intake, and adequate hydration before the procedure. However, given that there were doubts regarding the emergence of the left coronary artery and its pathway, coronary angiotomography was performed. The mass was found to surround the left main coronary artery, the proximal segment of the left anterior descending coronary artery, the circumflex coronary artery, and two septal arteries, as well as the great cardiac vein ( Figure 5, Figure 6, Figure 7 ). At that time, and because of these findings, the intervention was considered to be a high-risk procedure. Isotopic radiation with 131 I meta-iodine benzyl guanidine (MIBG) was considered as a palliative intervention, but this approach was rejected because of the risk for hypertensive crises of unpredictable severity. Another approach that was considered was the use of octeotride, but this alternative was not accepted by the multidisciplinary group, because there was a risk for irradiation of adjacent tissue, including the coronary arteries, with an unknown benefit. The risk/benefit assessment of this therapy did not favor the patient either, and surgery was thought to be the only option. Coronary angiography was done looking for a main feeding artery that could be embolized preoperatively to reduce the tumor size and intraoperative blood loss. However, embolization could not be done, because there were many small feeding arteries coming from the left main coronary artery ( Figure 8 ). The patient underwent mass resection, requiring a complete transverse arteriotomy of the ascending aorta, pulmonary trunk, and section of the superior vena cava. The mass was exposed using caudal traction and was found to be closely associated with the posterior wall of the ascending aorta, the pulmonary artery, and the left atrial roof, without infiltration of these structures. Involvement of the left coronary artery trunk was documented, which required ligation and revascularization of the anterior descending coronary artery and the circumflex coronary artery. The mass could be completely resected ( Figure 9 ). Figure 5 Coronary computed tomography. Mass surrounding the left main coronary artery and the proximal segment of the anterior descending and the circumflex coronary arteries. Figure 6 Coronary computed tomography. Mass surrounding the pulmonary artery. Figure 7 Coronary computed tomography. Mass located in the left atrioventricular groove. Figure 8 Coronary arteriography. Mass irrigated by multiple small vessels arising from the anterior descending coronary artery. Figure 9 Surgical specimen. Resected mass. The patient progressed well and was able to be extubated and have her vasopressor support discontinued. Follow-up echocardiography showed that systolic function was moderately affected, with an ejection fraction of 35% and diffuse hypokinesia, which was more pronounced in the anterior descending coronary artery territory. Because of these findings, new coronary angiography was performed, ruling out saphenous bridge compromise ( Figure 10 ). Further follow-up echocardiography showed improvement in systolic function, with an ejection fraction of 55% and preserved contractility of all segments except the basal segment of the anterior septum, which continued to be akinetic. There was also moderate pericardial effusion without increased intrapericardial pressure ( Figure 11, Videos 3 and 4 ). Figure 10 Coronary angiography. Saphenous bridge patent to the anterior descending artery, without lesions. Figure 11 Transthoracic echocardiography. (1) Parasternal view in diastole: pericardial effusion. (2) Parasternal view in systole: akinesia of the basal anteroseptal segment. The pathology report of the surgical specimen described round and oval cells in an organoid pattern with fine chromatin nuclei and degenerative focal atypia ( Figure 12 ). These findings and the immunohistochemical studies were compatible with a paraganglioma. The genetic study showed a succinate dehydrogenase subunit B mutation. The patient progressed satisfactorily and was discharged. One month later, it was found that one of her father's cousins had a para-aortic pheochromocytoma, an incidental finding in the emergency department due to abdominal pain. Figure 12 Round and oval cells, arranged in an organoid pattern, with degenerative focal atypia."}
Patient-History
History
[ "The patient was a 22 - year - old woman with a clinical history of recurrent episodes of palpitations, diaphoresis, and headache. She was found to have high blood pressure", "one of her father 's cousins had a para - aortic pheochromocytoma, an incidental finding in the emergency department due to abdominal pain" ]
test-400
6302035
{'Case Presentation': "We present a case of cardiac paraganglioma with left coronary artery involvement. The patient was a 22-year-old woman with a clinical history of recurrent episodes of palpitations, diaphoresis, and headache. She was found to have high blood pressure and, because of her age, several studies were done looking for secondary hypertension. Results of transthoracic echocardiography and renal Doppler imaging were normal, and plasma and urine metanephrine levels were within normal limits. Ambulatory blood pressure monitoring found severe hypertension, and the patient started treatment with angiotensin receptor blockers. After 2 years, she continued to have the same symptoms, but at this time, a mesocardial systolic murmur with back irradiation was found on auscultation. New transthoracic echocardiography showed high velocities in the pulmonary artery and a mass with intermediate echogenicity located at the base of the heart in close relation to the aorta and the pulmonary artery, extending into the left atrioventricular groove ( Figure 1, Videos 1 and 2 ). Free metanephrines in plasma (noradrenaline 4,886.67 pg/mL) and urine (noradrenaline 1,426.9 μg/24 h) were high. Figure 1 Transthoracic echocardiography. Shown is a mass ( white arrow ) with intermediate echogenicity in relation to the great vessels. To obtain better characterization, cardiac magnetic resonance was performed and showed a middle mediastinal mass located in the left atrioventricular groove, extending posteriorly to the left atrial roof and almost surrounding the pulmonary artery anteriorly. The mass was closely associated with the aortic root, but the emergence and path of the left coronary artery and its branches were not adequately visualized ( Figure 2 ). Tissue characterization sequences showed a mass with a heterogeneous appearance on T2-weighted sequences and slightly hyperintense on T1-weighted and T2 with fat saturation sequences ( Figure 2 ). Important mass vascularization was demonstrated with the perfusion sequence, which showed rapid uptake of contrast shortly after its administration. We also found focal late gadolinium enhancement at the periphery of the mass, with no enhancement at its center, a finding that was considered highly suggestive of a paraganglioma ( Figure 3 ). Fluorine-18 fluorodeoxyglucose positron emission tomography was also performed, demonstrating a highly metabolic mass ( Figure 4 ). Figure 2 Cardiac magnetic resonance. (1) Coronal: isointense mass in relation to the left atrial roof. (2) Two-chamber longitudinal: isointense mass in the left atrioventricular groove. (3,4) Axial: mass in relation to the aortic root and the pulmonary artery. Images reproduced with permission from Clínica Shaio-Bogotá-Colombia. Figure 3 Cardiac magnetic resonance. Delayed enhancement. Focal deposit of gadolinium in the periphery of the mass. Images reproduced with permission from Clínica Shaio-Bogotá-Colombia. Figure 4 Positron emission tomography with 18F-fluorodeoxyglucose (10 mCi). Hypermetabolic mass in the middle mediastinum. Images reproduced with permission from Clínica del Country-Bogotá-Colombia. The case was discussed with the heart team and other departments, such as endocrinology and oncology, concluding that the patient had a clear indication for surgery, with α- and β-blockade, high sodium intake, and adequate hydration before the procedure. However, given that there were doubts regarding the emergence of the left coronary artery and its pathway, coronary angiotomography was performed. The mass was found to surround the left main coronary artery, the proximal segment of the left anterior descending coronary artery, the circumflex coronary artery, and two septal arteries, as well as the great cardiac vein ( Figure 5, Figure 6, Figure 7 ). At that time, and because of these findings, the intervention was considered to be a high-risk procedure. Isotopic radiation with 131 I meta-iodine benzyl guanidine (MIBG) was considered as a palliative intervention, but this approach was rejected because of the risk for hypertensive crises of unpredictable severity. Another approach that was considered was the use of octeotride, but this alternative was not accepted by the multidisciplinary group, because there was a risk for irradiation of adjacent tissue, including the coronary arteries, with an unknown benefit. The risk/benefit assessment of this therapy did not favor the patient either, and surgery was thought to be the only option. Coronary angiography was done looking for a main feeding artery that could be embolized preoperatively to reduce the tumor size and intraoperative blood loss. However, embolization could not be done, because there were many small feeding arteries coming from the left main coronary artery ( Figure 8 ). The patient underwent mass resection, requiring a complete transverse arteriotomy of the ascending aorta, pulmonary trunk, and section of the superior vena cava. The mass was exposed using caudal traction and was found to be closely associated with the posterior wall of the ascending aorta, the pulmonary artery, and the left atrial roof, without infiltration of these structures. Involvement of the left coronary artery trunk was documented, which required ligation and revascularization of the anterior descending coronary artery and the circumflex coronary artery. The mass could be completely resected ( Figure 9 ). Figure 5 Coronary computed tomography. Mass surrounding the left main coronary artery and the proximal segment of the anterior descending and the circumflex coronary arteries. Figure 6 Coronary computed tomography. Mass surrounding the pulmonary artery. Figure 7 Coronary computed tomography. Mass located in the left atrioventricular groove. Figure 8 Coronary arteriography. Mass irrigated by multiple small vessels arising from the anterior descending coronary artery. Figure 9 Surgical specimen. Resected mass. The patient progressed well and was able to be extubated and have her vasopressor support discontinued. Follow-up echocardiography showed that systolic function was moderately affected, with an ejection fraction of 35% and diffuse hypokinesia, which was more pronounced in the anterior descending coronary artery territory. Because of these findings, new coronary angiography was performed, ruling out saphenous bridge compromise ( Figure 10 ). Further follow-up echocardiography showed improvement in systolic function, with an ejection fraction of 55% and preserved contractility of all segments except the basal segment of the anterior septum, which continued to be akinetic. There was also moderate pericardial effusion without increased intrapericardial pressure ( Figure 11, Videos 3 and 4 ). Figure 10 Coronary angiography. Saphenous bridge patent to the anterior descending artery, without lesions. Figure 11 Transthoracic echocardiography. (1) Parasternal view in diastole: pericardial effusion. (2) Parasternal view in systole: akinesia of the basal anteroseptal segment. The pathology report of the surgical specimen described round and oval cells in an organoid pattern with fine chromatin nuclei and degenerative focal atypia ( Figure 12 ). These findings and the immunohistochemical studies were compatible with a paraganglioma. The genetic study showed a succinate dehydrogenase subunit B mutation. The patient progressed satisfactorily and was discharged. One month later, it was found that one of her father's cousins had a para-aortic pheochromocytoma, an incidental finding in the emergency department due to abdominal pain. Figure 12 Round and oval cells, arranged in an organoid pattern, with degenerative focal atypia."}
{'Case Presentation': "We present a case of cardiac paraganglioma with left coronary artery involvement. The patient was a 22-year-old woman with a clinical history of recurrent episodes of palpitations, diaphoresis, and headache. She was found to have high blood pressure and, because of her age, several studies were done looking for secondary hypertension. Results of transthoracic echocardiography and renal Doppler imaging were normal, and plasma and urine metanephrine levels were within normal limits. Ambulatory blood pressure monitoring found severe hypertension, and the patient started treatment with angiotensin receptor blockers. After 2 years, she continued to have the same symptoms, but at this time, a mesocardial systolic murmur with back irradiation was found on auscultation. New transthoracic echocardiography showed high velocities in the pulmonary artery and a mass with intermediate echogenicity located at the base of the heart in close relation to the aorta and the pulmonary artery, extending into the left atrioventricular groove ( Figure 1, Videos 1 and 2 ). Free metanephrines in plasma (noradrenaline 4,886.67 pg/mL) and urine (noradrenaline 1,426.9 μg/24 h) were high. Figure 1 Transthoracic echocardiography. Shown is a mass ( white arrow ) with intermediate echogenicity in relation to the great vessels. To obtain better characterization, cardiac magnetic resonance was performed and showed a middle mediastinal mass located in the left atrioventricular groove, extending posteriorly to the left atrial roof and almost surrounding the pulmonary artery anteriorly. The mass was closely associated with the aortic root, but the emergence and path of the left coronary artery and its branches were not adequately visualized ( Figure 2 ). Tissue characterization sequences showed a mass with a heterogeneous appearance on T2-weighted sequences and slightly hyperintense on T1-weighted and T2 with fat saturation sequences ( Figure 2 ). Important mass vascularization was demonstrated with the perfusion sequence, which showed rapid uptake of contrast shortly after its administration. We also found focal late gadolinium enhancement at the periphery of the mass, with no enhancement at its center, a finding that was considered highly suggestive of a paraganglioma ( Figure 3 ). Fluorine-18 fluorodeoxyglucose positron emission tomography was also performed, demonstrating a highly metabolic mass ( Figure 4 ). Figure 2 Cardiac magnetic resonance. (1) Coronal: isointense mass in relation to the left atrial roof. (2) Two-chamber longitudinal: isointense mass in the left atrioventricular groove. (3,4) Axial: mass in relation to the aortic root and the pulmonary artery. Images reproduced with permission from Clínica Shaio-Bogotá-Colombia. Figure 3 Cardiac magnetic resonance. Delayed enhancement. Focal deposit of gadolinium in the periphery of the mass. Images reproduced with permission from Clínica Shaio-Bogotá-Colombia. Figure 4 Positron emission tomography with 18F-fluorodeoxyglucose (10 mCi). Hypermetabolic mass in the middle mediastinum. Images reproduced with permission from Clínica del Country-Bogotá-Colombia. The case was discussed with the heart team and other departments, such as endocrinology and oncology, concluding that the patient had a clear indication for surgery, with α- and β-blockade, high sodium intake, and adequate hydration before the procedure. However, given that there were doubts regarding the emergence of the left coronary artery and its pathway, coronary angiotomography was performed. The mass was found to surround the left main coronary artery, the proximal segment of the left anterior descending coronary artery, the circumflex coronary artery, and two septal arteries, as well as the great cardiac vein ( Figure 5, Figure 6, Figure 7 ). At that time, and because of these findings, the intervention was considered to be a high-risk procedure. Isotopic radiation with 131 I meta-iodine benzyl guanidine (MIBG) was considered as a palliative intervention, but this approach was rejected because of the risk for hypertensive crises of unpredictable severity. Another approach that was considered was the use of octeotride, but this alternative was not accepted by the multidisciplinary group, because there was a risk for irradiation of adjacent tissue, including the coronary arteries, with an unknown benefit. The risk/benefit assessment of this therapy did not favor the patient either, and surgery was thought to be the only option. Coronary angiography was done looking for a main feeding artery that could be embolized preoperatively to reduce the tumor size and intraoperative blood loss. However, embolization could not be done, because there were many small feeding arteries coming from the left main coronary artery ( Figure 8 ). The patient underwent mass resection, requiring a complete transverse arteriotomy of the ascending aorta, pulmonary trunk, and section of the superior vena cava. The mass was exposed using caudal traction and was found to be closely associated with the posterior wall of the ascending aorta, the pulmonary artery, and the left atrial roof, without infiltration of these structures. Involvement of the left coronary artery trunk was documented, which required ligation and revascularization of the anterior descending coronary artery and the circumflex coronary artery. The mass could be completely resected ( Figure 9 ). Figure 5 Coronary computed tomography. Mass surrounding the left main coronary artery and the proximal segment of the anterior descending and the circumflex coronary arteries. Figure 6 Coronary computed tomography. Mass surrounding the pulmonary artery. Figure 7 Coronary computed tomography. Mass located in the left atrioventricular groove. Figure 8 Coronary arteriography. Mass irrigated by multiple small vessels arising from the anterior descending coronary artery. Figure 9 Surgical specimen. Resected mass. The patient progressed well and was able to be extubated and have her vasopressor support discontinued. Follow-up echocardiography showed that systolic function was moderately affected, with an ejection fraction of 35% and diffuse hypokinesia, which was more pronounced in the anterior descending coronary artery territory. Because of these findings, new coronary angiography was performed, ruling out saphenous bridge compromise ( Figure 10 ). Further follow-up echocardiography showed improvement in systolic function, with an ejection fraction of 55% and preserved contractility of all segments except the basal segment of the anterior septum, which continued to be akinetic. There was also moderate pericardial effusion without increased intrapericardial pressure ( Figure 11, Videos 3 and 4 ). Figure 10 Coronary angiography. Saphenous bridge patent to the anterior descending artery, without lesions. Figure 11 Transthoracic echocardiography. (1) Parasternal view in diastole: pericardial effusion. (2) Parasternal view in systole: akinesia of the basal anteroseptal segment. The pathology report of the surgical specimen described round and oval cells in an organoid pattern with fine chromatin nuclei and degenerative focal atypia ( Figure 12 ). These findings and the immunohistochemical studies were compatible with a paraganglioma. The genetic study showed a succinate dehydrogenase subunit B mutation. The patient progressed satisfactorily and was discharged. One month later, it was found that one of her father's cousins had a para-aortic pheochromocytoma, an incidental finding in the emergency department due to abdominal pain. Figure 12 Round and oval cells, arranged in an organoid pattern, with degenerative focal atypia."}
Neurology
Neuro
[ "headache." ]