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prion
prion
[ "Gerstmann-Sträussler-Scheinker Syndrome (GSS)", "Genetic Creutzfeldt-Jakob Disease (Genetic CJD)", "Fatal Familial Insomnia (FFI)", "Major prion protein", "PRNP", "Genetic Prion Disease" ]
Genetic Prion Disease
Inga Zerr, Matthias Schmitz
Summary Genetic prion disease generally manifests with cognitive difficulties, ataxia, and myoclonus (abrupt jerking movements of muscle groups and/or entire limbs). The order of appearance and/or predominance of these features and other associated neurologic and psychiatric findings vary. The three major phenotypes of...
Genetic Creutzfeldt-Jakob Disease (genetic CJD) Fatal familial insomnia (FFI) Gerstmann-Sträussler-Scheinker syndrome (GSS) May also be referred to as familial CJD • Genetic Creutzfeldt-Jakob Disease (genetic CJD) • Fatal familial insomnia (FFI) • Gerstmann-Sträussler-Scheinker syndrome (GSS) ## Diagnosis Genet...
[]
27/3/2003
7/1/2021
7/9/2010
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
prolidase-def
prolidase-def
[ "Xaa-Pro dipeptidase", "PEPD", "Prolidase Deficiency" ]
Prolidase Deficiency
Francis Rossignol, Heng Wang, Carlos Ferreira
Summary Prolidase deficiency is characterized by skin lesions (typically severe, chronic, recalcitrant, and painful skin ulcers of the lower extremities and telangiectasias of the face and hands), recurrent infections (particularly of the skin and respiratory tract), dysmorphic facial features, variable intellectual di...
## Diagnosis No consensus clinical diagnostic criteria for prolidase deficiency have been published. Prolidase deficiency Skin lesions, typically lower-extremity ulcers ( Recurrent infections, particularly of the skin and respiratory tract Chronic lung disease with digital clubbing and a cystic fibrosis-like phe...
[]
25/6/2015
7/7/2022
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
prop1
prop1
[ "PROP1-Related CPHD", "PROP1-Related CPHD", "Homeobox protein prophet of Pit-1", "PROP1", "PROP1-Related Combined Pituitary Hormone Deficiency" ]
Luciani Renata Carvalho, Mirian Yumie Nishi, Fernanda Azevedo Correa, Juliana Moreira Marques, Ivo Jorge Prado Arnhold, Berenice B Mendonca
Summary Most affected individuals are ascertained because of short stature during childhood. Although TSH deficiency can present shortly after birth, TSH deficiency usually occurs with or after the onset of GH deficiency. Hypothyroidism is usually mild. FSH and LH deficiencies are typically identified at the age of ons...
## Diagnosis Thyroid-stimulating hormone (TSH) The two gonadotropins, luteinizing hormone (LH) and follicle-stimulating hormone (FSH) Prolactin (PrL) Adrenocorticotropic hormone (ACTH) (Deficiency develops in ~50% of individuals.) Delayed bone maturation on x-ray examination On head MRI, normal pituitary st...
[ "RV Araujo, CV Chang, VA Cescato, MC Fragoso, MD Bronstein, BB Mendonca, IJ Arnhold, LR Carvalho. PROP1 overexpression in corticotrophinomas: evidence for the role of PROP1 in the maintenance of cells committed to corticotrophic differentiation.. Clinics (Sao Paulo) 2013;68:887-91", "K Bajuk Studen, MA Stefanija,...
7/12/2000
24/2/2022
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
propionic-a
propionic-a
[ "Propionyl-CoA carboxylase alpha chain, mitochondrial", "Propionyl-CoA carboxylase beta chain, mitochondrial", "PCCA", "PCCB", "Propionic Acidemia" ]
Propionic Acidemia
Carolina I Galarreta Aima, Oleg A Shchelochkov, Teodoro Jerves Serrano, Charles P Venditti
Summary The spectrum of propionic acidemia (PA) ranges from neonatal onset to late-onset disease. Neonatal-onset PA, the most common form, is characterized by a healthy newborn with poor feeding and decreased arousal in the first few days of life, followed by progressive encephalopathy of unexplained origin. Without pr...
## Diagnosis NBS for propionic acidemia (PA) is primarily based on the quantification of propionylcarnitine (C3) and calculation of the C3/C2 ratio from the acylcarnitine profile on dried blood spots [ Propionylcarnitine (C3) and/or C3/C2 ratio values above the cutoff reported by the screening laboratory are consider...
[]
17/5/2012
26/9/2024
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
proteus
proteus
[ "AKT1-Related Overgrowth Spectrum", "AKT1-Related Proteus Syndrome", "RAC-alpha serine/threonine-protein kinase", "AKT1", "Proteus Syndrome" ]
Proteus Syndrome
Leslie G Biesecker, Julie C Sapp
Summary Proteus syndrome (PS) is characterized by progressive segmental or patchy overgrowth most commonly affecting the skeleton, skin, adipose, and central nervous systems. In most individuals PS has modest or no manifestations at birth, develops and progresses rapidly beginning in the toddler period, and relentlessl...
Proteus syndrome Proteus syndrome (clinical diagnosis) • Proteus syndrome • Proteus syndrome (clinical diagnosis) • Proteus syndrome (clinical diagnosis) • Proteus syndrome (clinical diagnosis) ## Diagnosis Consensus clinical diagnostic criteria for Proteus syndrome (PS) have been published [ PS Distorting, pr...
[ "K Abell, L Tolusso, N Smith, R Hopkin, M Vawter-Lee, M Habli, S Riddle, MA Calvo-Garcia, Q Guan, K Bierbrauer, V Hwa, HM Saal. Prenatal diagnosis of Proteus syndrome: diagnosis of an AKT1 mutation from amniocytes.. Birth Defects Res. 2020;112:1733-7", "T AlAnzi, E Al-Mashharawi, A Alhashem. Proteus syndrome caus...
9/8/2012
25/5/2023
10/1/2019
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
prrt2-parox
prrt2-parox
[ "PRRT2-Related Paroxysmal Kinesigenic Dyskinesia (PKD)", "PRRT2-Related Paroxysmal Kinesigenic Dyskinesia with Infantile Convulsions (PKD/IC)", "PRRT2-Related Hemiplegic Migraine", "PRRT2-Related Self-Limited (Familial) Infantile Epilepsy (SeLIE)", "Proline-rich transmembrane protein 2", "PRRT2", "PRRT2...
Kathryn Yang, Vincente Quiroz, Darius Ebrahimi-Fakhari
Summary The diagnosis of
## Diagnosis SeLIE is characterized by the following clinical and supportive findings [ Onset in first year of life (usually age 4-7 months) Spontaneous or in context of fever Occurring in clusters of multiple brief seizures per day: on average up to eight to ten seizures per day every two to three hours Excellent...
[]
11/1/2018
4/7/2024
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
prs
prs
[ "PRS Superactivity", "Ribose-phosphate pyrophosphokinase 1", "PRPS1", "Phosphoribosylpyrophosphate Synthetase Superactivity" ]
Phosphoribosylpyrophosphate Synthetase Superactivity
Arjan PM de Brouwer, John Christodoulou
Summary Phosphoribosylpyrophosphate synthetase (PRS) superactivity comprises two phenotypes, both characterized by hyperuricemia and hyperuricosuria. The mild phenotype (~75% of affected males) with onset in the second or third decade of life is typically limited to these biochemical findings, whereas the severe phenot...
Phosphoribosylpyrophosphate Synthetase (PRS) Superactivity: Phenotypes in Males and Females In all males with mild PRS superactivity evaluated to date, the sequence of the Biochemical testing is unlikely to be informative in asymptomatic females. ## Diagnosis No consensus clinical diagnostic criteria for phosphorib...
[ "MA Becker, M Kim, K Husain, T Kang. Regulation of purine nucleotide synthesis in human B lymphoblasts with both hypoxanthine-guanine phosphoribosyltransferase and phosphoribosylpyrophosphate synthetase superactivity.. J Biol Chem 1992;267:4317-21", "MA Becker, MJ Losman, M Kim. Mechanisms of accelerated purine n...
23/9/2008
17/2/2022
11/1/2011
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
prss1-hp
prss1-hp
[ "Serine protease 1", "PRSS1", "PRSS1-Related Hereditary Pancreatitis" ]
David C Whitcomb
Summary The diagnosis of High-penetrance HP caused by gain-of-function
## Diagnosis The clinical features of Acute pancreatitis occurring in childhood Recurrent acute attacks of pancreatitis of unknown cause Chronic pancreatitis of unknown cause, particularly with onset before age 25 years A family history of recurrent acute pancreatitis, chronic pancreatitis, and/or childhood pancre...
[]
1/3/2012
27/3/2025
GeneReviews®
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[ "Review", "Clinical Review" ]
psach
psach
[ "Pseudoachondroplasia (PSACH)", "Pseudoachondroplasia (PSACH)", "Cartilage oligomeric matrix protein", "COMP", "COMP-Related Pseudoachondroplasia" ]
Michael D Briggs, Michael J Wright
Summary The diagnosis of
## Diagnosis Normal length at birth Normal facies Waddling gait, recognized at the onset of walking Decline in growth rate to below the standard growth curve by approximately age two years, leading to moderately severe disproportionate short-limb short stature Moderate brachydactyly Ligamentous laxity and joint...
[]
20/8/2004
30/11/2023
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
ptps-def
ptps-def
[ "6-Pyruvoyl-Tetrahydropterin Synthase Deficiency", "PTS Deficiency", "6-Pyruvoyl-Tetrahydropterin Synthase Deficiency", "PTS Deficiency", "6-pyruvoyl tetrahydrobiopterin synthase", "PTS", "PTS-Related Tetrahydrobiopterin Deficiency" ]
Thomas Opladen, Nicola Longo, Nenad Blau
Summary In the mild (peripheral) form, affected individuals are usually asymptomatic apart from an increase in phenylalanine (Phe) levels. Some remain asymptomatic. However, with time, some have mild developmental delays and can develop deficiency of neurotransmitter production, such that treatment of some asymptomatic...
## Diagnosis Deficiency of tetrahydrobiopterin (BH4), a cofactor for the enzyme phenylalanine hydroxylase (PAH), can lead to accumulation of phenylalanine (Phe) in the blood (hyperphenylalaninemia). In addition, it impairs activity of tyrosine (Tyr) and tryptophan hydroxylase, leading to defective synthesis of neurotr...
[]
10/7/2025
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
ptt
ptt
[ "Prothrombin G20210A Thrombophilia", "Prothrombin G20210A Thrombophilia", "Prothrombin", "F2", "Prothrombin Thrombophilia" ]
Prothrombin Thrombophilia
Jody L Kujovich
Summary Prothrombin thrombophilia is characterized by venous thromboembolism (VTE) manifest most commonly in adults as deep-vein thrombosis (DVT) in the legs or pulmonary embolism. The clinical expression of prothrombin thrombophilia is variable; many individuals heterozygous or homozygous for the 20210G>A The diagnosi...
## Diagnosis No clinical features are specific for prothrombin thrombophilia. The diagnosis A first unprovoked venous thromboembolism (VTE) before age 50 years A history of recurrent VTE Venous thrombosis at certain unusual sites such as the cerebral, mesenteric, portal, or hepatic veins VTE during pregnancy or th...
[ "Practice Bulletin No.138: Inherited thrombophilias in pregnancy.. Obstet Gynecol. 2013;122:706-17" ]
25/7/2006
4/2/2021
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
pura-dis
pura-dis
[ "PURA Syndrome", "5q31.3 Deletion Syndrome", "Transcriptional activator protein Pur-alpha", "PURA", "PURA-Related Neurodevelopmental Disorders" ]
Margot RF Reijnders, Richard J Leventer, Bo Hoon Lee, Diana Baralle, Paulo Selber, Alex R Paciorkowski, David Hunt
Summary The diagnosis of a
5q31.3 deletion syndrome For synonyms and outdated names see • 5q31.3 deletion syndrome ## Diagnosis No formal clinical diagnostic criteria have been published for Hypotonia Neonatal hypoventilation Hypothermia Hypersomnolence Feeding difficulties, including gastroesophageal reflux disease (GERD) Hypotoni...
[ "MC Bonaglia, N Zanotta, R Giorda, G D'Angelo, C Zucca. Long-term follow-up of a patient with 5q31.3 microdeletion syndrome and the smallest de novo 5q31.2q31.3 deletion involving PURA.. Mol Cytogenet 2015;8:89", "N Brown, T Burgess, R Forbes, G McGillivray, A Kornberg, S Mandelstam, Z. Starl. 5q31.3 Microdeletio...
27/4/2017
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
pws
pws
[ "Prader-Labhart-Willi Syndrome", "Prader-Labhart-Willi Syndrome", "Prader-Willi Syndrome" ]
Prader-Willi Syndrome
Daniel J Driscoll, Jennifer L Miller, Suzanne B Cassidy
Summary Prader-Willi syndrome (PWS) is characterized by severe hypotonia, poor appetite, and feeding difficulties in early infancy, followed in early childhood by excessive eating and gradual development of morbid obesity (unless food intake is strictly controlled). Motor milestones and language development are delayed...
## Diagnosis Prader-Willi syndrome (PWS) Clinical findings differ by age group. The presence of Hypotonia with poor appetite and suck in the neonatal period Developmental delay Hypotonia with history of poor suck Developmental delay History of hypotonia with poor suck (hypotonia often persists) Developmen...
[]
6/10/1998
9/3/2023
5/12/2024
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
pxe
pxe
[ "PXE", "PXE", "ATP-binding cassette sub-family C member 6", "ABCC6", "Pseudoxanthoma Elasticum" ]
Pseudoxanthoma Elasticum
Sharon F Terry, Jouni Uitto
Summary Pseudoxanthoma elasticum (PXE) is a systemic disorder that affects the elastic tissue of the skin, the eye, and vascular system. Individuals most commonly present with angioid streaks of the retina found on routine eye examination or associated with retinal hemorrhage and/or characteristic papules in the skin. ...
## Diagnosis Formal diagnostic criteria for pseudoxanthoma elasticum (PXE) have been established [ PXE Papules (darker than the skin color), usually seen on the lateral aspect of the neck or the flexural creases, such as the antecubital fossae, axillae, groin, or popliteal fossae Plaques formed by coalescence o...
[ "JL Anderson, JL Halperin, NM Albert, B Bozkurt, RG Brindis, LH Curtis, D DeMets, RA Guyton, JS Hochman, RJ Kovacs, EM Ohman, SJ Pressler, FW Sellke, WK Shen. Management of patients with peripheral artery disease (compilation of 2005 and 2011 ACCF/AHA guideline recommendations): a report of the American College of ...
5/6/2001
4/6/2020
2/4/2007
GeneReviews®
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[ "Review", "Clinical Review" ]
pycnodys
pycnodys
[ "Pyknodysostosis", "Toulouse-Lautrec Syndrome", "CTSK-Related Pyknodysostosis", "Pyknodysostosis", "Toulouse-Lautrec Syndrome", "CTSK-Related Pyknodysostosis", "Cathepsin K", "CTSK", "Pycnodysostosis" ]
Pycnodysostosis
Shannon LeBlanc, Ravi Savarirayan
Summary Pycnodysostosis is characterized by short-limbed short stature, typical facial appearance (convex nasal ridge and small jaw with obtuse mandibular angle), osteosclerosis with increased bone fragility, acroosteolysis of the distal phalanges, delayed closure of the cranial sutures, and dysplasia of the clavicle. ...
## Diagnosis Formal diagnostic criteria for pycnodysostosis have not been established, however the radiographic features of acroosteolysis, osteosclerosis, and loss of the normal angle of the jaw are almost pathognomonic. Pycnodysostosis Short-limbed short stature in all individuals (prenatal onset in ~30%) Brach...
[ "NM Appelman-Dijkstra, SE Papapoulos. From disease to treatment: from rare skeletal disorders to treatments for osteoporosis.. Endocrine. 2016;52:414-26", "A Arman, A Bereket, A Coker, P Özlem, S Kiper, T Güran, B Özkan, Z Atay, T Akçay, B Haliloglu, K Boduroglu, Y Alanay, S. Turan. Cathepsin K analysis in a pych...
5/11/2020
6/4/2023
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
rab18-def
rab18-def
[ "Martsolf Syndrome", "Warburg Micro Syndrome", "Rab3 GTPase-activating protein catalytic subunit", "Rab3 GTPase-activating protein non-catalytic subunit", "Ras-related protein Rab-18", "TBC1 domain family member 20", "RAB18", "RAB3GAP1", "RAB3GAP2", "TBC1D20", "RAB18 Deficiency" ]
RAB18 Deficiency
Mark Handley, Eamonn Sheridan
Summary RAB18 deficiency is the molecular deficit underlying both Warburg micro syndrome (characterized by eye, nervous system, and endocrine abnormalities) and Martsolf syndrome (characterized by similar – but milder – findings). To date Warburg micro syndrome comprises >96% of reported individuals with genetically de...
Warburg micro syndrome Martsolf syndrome For synonyms and outdated names see For other genetic causes of these phenotypes see • Warburg micro syndrome • Martsolf syndrome ## Diagnosis RAB18 deficiency Note: Findings indicated with an * are the basis of the diagnosis when molecular genetic testing either has not...
[ "GM Abdel-Salam, NA Hassan, HF Kayed, IA Aligianis. Phenotypic variability in Micro syndrome: report of new cases.. Genet Couns. 2007;18:423-35", "IA Aligianis, CA Johnson, P Gissen, D Chen, D Hampshire, K Hoffmann, EN Maina, NV Morgan, L Tee, J Morton, JR Ainsworth, D Horn, E Rosser, TR Cole, I Stolte-Dijkstra, ...
4/1/2018
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
rapid-odp
rapid-odp
[ "Alternating Hemiplegia of Childhood (AHC)", "Cerebellar Ataxia, Areflexia, Pes Cavus, Optic Atrophy, and Sensorineural Hearing Loss (CAPOS) Syndrome", "Rapid-Onset Dystonia-Parkinsonism (RDP)", "Relapsing Encephalopathy with Cerebellar Ataxia (RECA) / Fever-Induced Paroxysmal Weakness and Encephalopathy (FIP...
Allison Brashear, Kathleen J Sweadner, Ihtsham Haq, Eleonora Napoli, Laurie Ozelius
Summary AHC is characterized by onset prior to age 18 months of paroxysmal hemiplegic episodes, predominately involving the limbs and/or the whole body, lasting from minutes to hours to days (and sometimes weeks) with remission only during sleep, only to resume after awakening. Although paroxysmal episodic neurologic d...
Alternating hemiplegia of childhood (AHC) Cerebellar ataxia, areflexia, pes cavus, optic atrophy, sensorineural hearing loss (CAPOS) syndrome Relapsing encephalopathy with cerebellar ataxia (RECA) / fever-induced paroxysmal weakness and encephalopathy (FIPWE) Rapid-onset dystonia-parkinsonism (RDP) For synonyms and...
[]
7/2/2008
5/12/2024
13/9/2012
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
rasa1-rel-dis
rasa1-rel-dis
[ "CM-AVM Syndrome", "CM-AVM Syndrome", "RASA1 Parkes Weber Syndrome", "Ephrin type-B receptor 4", "Ras GTPase-activating protein 1", "EPHB4", "RASA1", "Capillary Malformation-Arteriovenous Malformation Syndrome" ]
Capillary Malformation-Arteriovenous Malformation Syndrome
Pinar Bayrak-Toydemir, David A Stevenson
Summary Capillary malformation-arteriovenous malformation (CM-AVM) syndrome is characterized by the presence of multiple small (1-2 cm in diameter) capillary malformations mostly localized on the face and limbs. Some affected individuals also have associated arteriovenous malformations (AVMs) and/or arteriovenous fistu...
For synonyms and outdated names see For other genetic causes of this phenotype see ## Diagnosis Diagnostic criteria for capillary malformation-arteriovenous malformation (CM-AVM) syndrome have been proposed but not systematically evaluated [ CM-AVM syndrome Multifocal, atypical pink-to-reddish brown, multiple, sma...
[]
22/2/2011
12/9/2019
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
rbs
rbs
[ "Roberts Syndrome", "SC Phocomelia Syndrome", "N-acetyltransferase ESCO2", "ESCO2", "ESCO2 Spectrum Disorder" ]
Hugo Vega, Miriam Gordillo, Ethylin Wang Jabs
Summary The diagnosis of
Roberts syndrome SC phocomelia syndrome For synonyms and outdated names see • Roberts syndrome • SC phocomelia syndrome ## Diagnosis The diagnosis Note: (1) Per American College of Medical Genetics and Genomics / Association for Molecular Pathology variant interpretation guidelines, the terms "pathogenic var...
[]
18/4/2006
14/8/2025
14/4/2009
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
rcdp
rcdp
[ "PEX7-Rhizomelic CDP", "Rhizomelic Chondrodysplasia Punctata Type 1", "Rhizomelic Chondrodysplasia Punctata Type 1", "PEX7-Rhizomelic CDP", "Peroxisomal targeting signal 2 receptor", "PEX7", "PEX7-Related Rhizomelic Chondrodysplasia Punctata" ]
Nancy E Braverman, Ricki Carroll, Wedad Fallatah, Mahim Jain
Summary The diagnosis of
## Diagnosis Congenital cataracts Skeletal/radiographic findings Rhizomelia (proximal shortening of the long bones) Chondrodysplasia punctata (CDP). Punctate calcifications observed in radiographs in the epiphyseal cartilage at the knee, hip, elbow, and shoulder that can be more extensive, involving the hyoid bon...
[]
16/11/2001
7/8/2025
7/2/2005
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
refsum
refsum
[ "Classic Refsum Disease", "Classic Refsum Disease", "Peroxisomal targeting signal 2 receptor", "Phytanoyl-CoA dioxygenase, peroxisomal", "PEX7", "PHYH", "Adult Refsum Disease" ]
Adult Refsum Disease
Hans R Waterham, Ronald JA Wanders, Bart P Leroy
Summary Adult Refsum disease (ARD) is associated with elevated plasma phytanic acid levels, late childhood-onset (or later) retinitis pigmentosa, and variable combinations of anosmia, polyneuropathy, deafness, ataxia, and ichthyosis. Onset of symptoms ranges from age seven months to older than age 50 years. Cardiac arr...
## Diagnosis Adult Refsum disease (ARD), also referred to as "classic Refsum disease," is a peroxisomal disorder. In the majority of individuals, it is caused by a deficiency of the peroxisomal enzyme phytanoyl-CoA hydroxylase due to biallelic pathogenic variants in No consensus clinical diagnostic criteria for ARD h...
[]
20/3/2006
30/9/2021
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
rere-dis
rere-dis
[ "Neurodevelopmental Disorder with or without Anomalies of the Brain, Eye, or Heart (NEDBEH)", "Neurodevelopmental Disorder with or without Anomalies of the Brain, Eye, or Heart (NEDBEH)", "Arginine-glutamic acid dipeptide repeats protein", "RERE", "RERE-Related Disorders" ]
Daryl A Scott, Elliott H Sherr
Summary The diagnosis of
## Diagnosis No clinical diagnostic criteria have been published. Behavioral issues, including attention-deficit/hyperactivity disorder and self-injurious behavior Generalized hypotonia of infancy Infant feeding difficulties Eye/vision problems: Structural eye defects (coloboma, optic nerve atrophy/hypoplasia, mi...
[ "B Fregeau, BJ Kim, A Hernández-García, VK Jordan, MT Cho, RE Schnur, KG Monaghan, J Juusola, JA Rosenfeld, E Bhoj, EH Zackai, S Sacharow, K Barañano, DGM Bosch, BBA de Vries, K Lindstrom, A Schroeder, P James, P Kulch, SR Lalani, MM van Haelst, KLI van Gassen, E van Binsbergen, AJ Barkovich, DA Scott, EH Sherr. De...
21/3/2019
GeneReviews®
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[ "Review", "Clinical Review" ]
retinoblastoma
retinoblastoma
[ "Retinoblastoma-associated protein", "RB1", "Retinoblastoma" ]
Retinoblastoma
Dietmar R Lohmann, Brenda L Gallie
Summary Retinoblastoma is a malignant tumor of the developing retina that occurs in children, usually before age five years. Retinoblastoma may be unifocal or multifocal. About 60% of affected individuals have unilateral retinoblastoma with a mean age of diagnosis of 24 months; about 40% have bilateral retinoblastoma w...
## Diagnosis Guidelines for diagnosis and care of children and families affected by retinoblastoma have been published [ Leukocoria (white pupil) Strabismus Change in eye appearance Reduced visual acuity Diagnosis of retinoblastoma, including unilateral (unifocal and multifocal) and bilateral involvement Retinom...
[]
18/7/2000
21/9/2023
GeneReviews®
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[ "Review", "Clinical Review" ]
retinoschisis
retinoschisis
[ "X-Linked Retinoschisis", "X-Linked Retinoschisis", "Retinoschisin", "RS1", "X-Linked Congenital Retinoschisis" ]
X-Linked Congenital Retinoschisis
Paul A Sieving, Ian M MacDonald, Stephanie Hoang
Summary X-linked congenital retinoschisis (XLRS) is characterized by symmetric bilateral macular involvement with onset in the first decade of life, in some cases as early as age three months. Fundus examination shows areas of schisis (splitting of the nerve fiber layer of the retina) in the macula, sometimes giving th...
## Diagnosis X-linked congenital retinoschisis (XLRS) Bilaterally reduced visual acuity, typically between 20/60 and 20/120 No presenting complaint of “night blindness” (i.e., vision difficulty in dim lighting) Fundus examination revealing: Areas of schisis (cavities and splitting through the deeper retinal laye...
[]
24/10/2003
5/11/2020
18/6/2007
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
rett
rett
[ "MECP2 Classic Rett Syndrome", "MECP2-Related Severe Neonatal Encephalopathy", "Pyramidal Signs, Parkinsonism, and Macroorchidism (PPM-X) Syndrome", "Variant Rett Syndrome", "Methyl-CpG-binding protein 2", "MECP2", "MECP2 Disorders" ]
Simranpreet Kaur, John Christodoulou
Summary The spectrum of The diagnosis of a
Variant Rett syndrome Mild learning disabilities Pyramidal signs, parkinsonism, and macroorchidism (PPM-X) syndrome Syndromic/nonsyndromic intellectual disability For other genetic causes of these phenotypes see Note: The allelic disorder • Variant Rett syndrome • Mild learning disabilities • Pyramidal signs, p...
[]
3/10/2001
19/9/2019
GeneReviews®
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[ "Review", "Clinical Review" ]
rfc1-canvas
rfc1-canvas
[ "RFC1-CANVAS", "RFC1-Cerebellar Ataxia, Neuropathy, Vestibular Areflexia Syndrome", "Replication factor C subunit 1", "RFC1", "RFC1 CANVAS / Spectrum Disorder" ]
Andrea Cortese, Mary M Reilly, Henry Houlden
Summary The phenotypic spectrum associated with biallelic The diagnosis of
The phenotypic spectrum associated with biallelic intronic AAGGG pentanucleotide expansions in ## Diagnosis Formal diagnostic criteria for Symptoms include unsteadiness (imbalance, dizziness), falls, clumsiness of hands. Examination reveals progressive ataxia of gait and limb dysmetria. Symptoms include unstea...
[ "D Aboud Syriani, D Wong, S Andani, CM De Gusmao, Y Mao, M Sanyoura, G Glotzer, PJ Lockhart, S Hassin-Baer, V Khurana, CM Gomez, S Perlman, S Das, BL Fogel. Prevalence of RFC1-mediated spinocerebellar ataxia in a North American ataxia cohort.. Neurol Genet 2020;6", "F Akçimen, JP Ross, V. Cynthia, CV Bourassa, C ...
25/11/2020
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
riboflavin-tn
riboflavin-tn
[ "Solute carrier family 52, riboflavin transporter, member 1", "Solute carrier family 52, riboflavin transporter, member 2", "Solute carrier family 52, riboflavin transporter, member 3", "SLC52A1", "SLC52A2", "SLC52A3", "Riboflavin Transporter Deficiency" ]
Riboflavin Transporter Deficiency
Elisa Cali, Natalia Dominik, Andreea Manole, Henry Houlden
Summary Riboflavin transporter deficiency (RTD), comprising RTD2 and RTD3 (caused by biallelic pathogenic variants in In the majority of affected individuals, the initial finding is sensorineural hearing loss, which is usually progressive and severe. The time between the onset of hearing loss and the development of oth...
Riboflavin transporter deficiency (RTD), comprising RTD2 and RTD3 (caused by biallelic pathogenic variants in One case report (which requires additional confirmation) suggests that biallelic expression of pathogenic variants in ## Diagnosis Very frequently affecting cranial nerves II (optic atrophy with bilateral sy...
[ "Q Abbas, SK Jafri, S Ishaque, AJ Rahman. Brown-Vialetto-Van Laere syndrome: a novel diagnosis to a common presentation.. BMJ Case Rep. 2018;2018", "F Amir, C Atzinger, K Massey, J Greinwald, LL Hunter, E Ulm, M Kettler. The clinical journey of patients with riboflavin transporter deficiency type 2.. J Child Neur...
11/6/2015
8/4/2021
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
rickets-xlh
rickets-xlh
[ "X-Linked Hypophosphatemic Rickets (XLHR)", "X-Linked Vitamin D-Resistant Rickets", "Hypophosphatemic Rickets, PHEX-Related", "X-Linked Vitamin D-Resistant Rickets", "X-Linked Hypophosphatemic Rickets (XLHR)", "Hypophosphatemic Rickets, PHEX-Related", "Phosphate-regulating neutral endopeptidase PHEX", ...
X-Linked Hypophosphatemia
Michaël R Laurent, Pol Harvengt, Geert R Mortier, Detlef Böckenhauer
Summary The phenotypic spectrum of X-linked hypophosphatemia (XLH) ranges from isolated hypophosphatemia to severe lower extremity bowing and/or craniosynostosis, usually involving the sagittal suture with consequent scaphocephaly. XLH typically manifests in the first two years of life with lower extremity bowing due t...
## Diagnosis For the purposes of this X-linked hypophosphatemia (XLH) Clinical signs of rickets resistant to treatment with regular vitamin D Progressive lower extremity bowing Decrease in height velocity after the child starts ambulating Epiphyseal swelling Harrison groove (a horizontal channel at the lower e...
[]
9/2/2012
14/12/2023
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
ritscher-schinzel
ritscher-schinzel
[ "3C Syndrome", "Cranio-Cerebello-Cardiac Dysplasia", "3C Syndrome", "Cranio-Cerebello-Cardiac Dysplasia", "Coiled-coil domain-containing protein 22", "WASH complex subunit 5", "CCDC22", "WASHC5", "Ritscher-Schinzel Syndrome" ]
Ritscher-Schinzel Syndrome
Alison M Elliott, Albert Chudley
Summary Ritscher-Schinzel syndrome (RSS) is a clinically recognizable condition that includes the cardinal findings of craniofacial features, cerebellar defects, and cardiovascular malformations resulting in the alternate diagnostic name of 3C syndrome. Dysmorphic facial features may include brachycephaly, hypotonic fa...
## Diagnosis Consensus clinical diagnostic criteria for Ritscher-Schinzel syndrome (RSS) have not been established. Congenital heart malformation(s) other than patent ductus arteriosus alone Dandy-Walker malformation, cerebellar vermis hypoplasia, or enlarged cisterna magna Cleft palate OR ocular coloboma OR four o...
[]
23/1/2020
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
rnu4atac-dis
rnu4atac-dis
[ "RNU4ATAC Spectrum Disorder", "RNU4ATAC Spectrum Disorder", "Microcephalic Osteodysplastic Primordial Dwarfism Type I/III (MOPDI) / Taybi-Linder Syndrome", "Roifman Syndrome", "Lowry-Wood Syndrome", "N/A (non-coding RNA)", "RNU4ATAC", "RNU4atac-opathy" ]
RNU4atac-opathy
Angela Duker, Danita Velasco, Nic Robertson, Andrew Jackson, Magee DeFelice, Michael B Bober
Summary RNU4atac-opathy encompasses the phenotypic spectrum of biallelic The diagnosis of RNU4atac-opathy is established in a proband with suggestive findings and biallelic pathogenic (or likely pathogenic) variants in RNU4atac-opathy is inherited in an autosomal recessive manner. If both parents are known to be hetero...
With the current widespread use of multigene panels and comprehensive genomic testing, it has become apparent that the phenotypic spectrum of biallelic The need to evaluate an individual found to have The importance of counseling families that the finding of biallelic RNU4atac-opathy: Phenotypic Spectrum Associated ...
[]
16/2/2023
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
rob-ad
rob-ad
[ "Fetal Face Syndrome", "Fetal Face Syndrome", "Protein Wnt-5a", "Segment polarity protein dishevelled homolog DVL-1", "Segment polarity protein dishevelled homolog DVL-3", "DVL1", "DVL3", "WNT5A", "Autosomal Dominant Robinow Syndrome" ]
Autosomal Dominant Robinow Syndrome
Maian Roifman, Han Brunner, Jamie Lohr, Juliana Mazzeu, David Chitayat
Summary Autosomal dominant Robinow syndrome (ADRS) is characterized by skeletal findings (short stature, mesomelic limb shortening predominantly of the upper limbs, and brachydactyly), genital abnormalities (in males: micropenis / webbed penis, hypoplastic scrotum, cryptorchidism; in females: hypoplastic clitoris and l...
## Diagnosis Autosomal dominant Robinow syndrome (ADRS) Short stature Mesomelic limb shortening predominantly affecting the upper limbs Brachydactyly In males: micropenis / webbed penis, hypoplastic scrotum, and cryptorchidism In females: hypoplastic clitoris and labia majora Dysmorphic facial features res...
[ "J Al-Ata, M Paquet, AS Teebi. Congenital heart disease in Robinow syndrome.. Am J Med Genet. 1998;77:332-3", "S Beiraghi, V Leon-Salazar, BE Larson, MT John, ML Cunningham, A Petryk, JL Lohr. Craniofacial and intraoral phenotype of Robinow syndrome forms.. Clin Genet. 2011;80:15-24", "KJ Bunn, P Daniel, HS Ros...
8/1/2015
9/8/2018
3/10/2019
GeneReviews®
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[ "Review", "Clinical Review" ]
rob
rob
[ "Fetal Face Syndrome", "Fetal Face Syndrome", "Tyrosine-protein kinase transmembrane receptor ROR2", "ROR2", "ROR2-Related Robinow Syndrome" ]
Carlos A Bacino
Summary The diagnosis of
## Diagnosis Macrocephaly Dysmorphic facial features including: broad prominent forehead, marked ocular hypertelorism, prominent eyes with apparent exophthalmos resulting from deficiency of the lower eyelid (giving the eyes a more prominent appearance), midface hypoplasia, short upturned nose with depressed nasal bri...
[ "AR Afzal, S Jeffery. One gene, two phenotypes: ROR2 mutations in autosomal recessive Robinow syndrome and autosomal dominant brachydactyly type B.. Hum Mutat. 2003;22:1-11", "AR Afzal, A Rajab, CD Fenske, M Oldridge, N Elanko, E Ternes-Pereira, B Tuysuz, VA Murday, MA Patton, AO Wilkie, S Jeffery. Recessive Robi...
28/7/2005
26/7/2018
12/9/2019
GeneReviews®
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[ "Review", "Clinical Review" ]
rotor
rotor
[ "Rotor-Type Hyperbilirubinemia", "Rotor-Type Hyperbilirubinemia", "Solute carrier organic anion transporter family member 1B1", "Solute carrier organic anion transporter family member 1B3", "SLCO1B1", "SLCO1B3", "Rotor Syndrome" ]
Rotor Syndrome
Milan Jirsa, AS Knisely, Alfred Schinkel, Stanislav Kmoch
Summary Rotor syndrome is characterized by mild conjugated and unconjugated hyperbilirubinemia that usually begins shortly after birth or in childhood. Jaundice may be intermittent. Conjunctival icterus may be the only clinical manifestation. The diagnosis of Rotor syndrome is established in a proband with isolated, pr...
## Diagnosis Rotor syndrome Mild jaundice (may be intermittent) Conjunctival icterus (in some affected individuals) Otherwise normal physical examination Conjugated hyperbilirubinemia with serum total bilirubin concentration usually between 2 and 5 mg/dL but possibly higher. Conjugated bilirubin usually exceeds ...
[]
13/12/2012
27/2/2025
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
rp-overview
rp-overview
[ "Adhesion G protein-coupled receptor A3", "ADP-ribosylation factor-like protein 2-binding protein", "ADP-ribosylation factor-like protein 3", "ADP-ribosylation factor-like protein 6", "BBSome complex member BBS1", "BBSome complex member BBS2", "Bestrophin-1", "Carbonic anhydrase 4", "CCA tRNA nucleo...
Nonsyndromic Retinitis Pigmentosa Overview
Abigail T Fahim, Stephen P Daiger, Richard G Weleber
Summary The purpose of this overview is to: Describe the Review the Provide an Provide a brief summary of Inform
## Clinical Characteristics of Nonsyndromic Retinitis Pigmentosa Retinitis pigmentosa (RP) refers to a group of inherited disorders in which abnormalities of the photoreceptors (rods and cones) of the retina lead to progressive visual loss. RP is classified as nonsyndromic, or "simple" (not affecting other organs or ...
[]
4/8/2000
19/1/2017
6/4/2023
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
rpe65-lca
rpe65-lca
[ "RPE65-Related LCA/EOSRD", "RPE65-Related LCA / EOSRD", "Retinoid isomerohydrolase", "RPE65", "RPE65-Related Leber Congenital Amaurosis / Early-Onset Severe Retinal Dystrophy" ]
Daniel L Chao, Amanda Burr, Mark Pennesi
Summary The diagnosis of Subretinal gene augmentation, an FDA-approved therapy, compensates for loss-of-function
## Diagnosis Symptomatic onset between birth and age five years Roving eye movements or nystagmus Poor pupillary light responses in some Profound nyctalopia Oculodigital sign (i.e., poking, rubbing or pressing on the eye in order to stimulate phosphenes for visual perception). Once considered pathognomonic for L...
[ "L Al-Gazali, BR Ali. Mutations of a country: a mutation review of single gene disorders in the United Arab Emirates (UAE).. Hum Mutat. 2010;31:505-20", "GDN Astuti, M Bertelsen, MN Preising, M Ajmal, B Lorenz, SMH Faradz, R Qamar, RWJ Collin, T Rosenberg, FPM Cremers. Comprehensive genotyping reveals RPE65 as th...
14/11/2019
GeneReviews®
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[ "Review", "Clinical Review" ]
rrm2b-mtddepl
rrm2b-mtddepl
[ "RRM2B Autosomal Recessive Progressive External Ophthalmoplegia (arPEO)", "RRM2B Encephalomyopathic Mitochondrial DNA Maintenance Defects (MDMD)", "RRM2B Mitochondrial Neurogastrointestinal Encephalopathy (MNGIE)", "RRM2B Autosomal Dominant Progressive External Ophthalmoplegia (adPEO)", "Ribonucleoside-diph...
Albert Z Lim, Robert McFarland, Robert W Taylor, Gráinne S Gorman
Summary Four phenotypes comprise the To date, 78 individuals from 52 families with a molecularly confirmed The diagnosis of an With the exception of autosomal dominant progressive external ophthalmoplegia, Once the
COX = cytochrome No true epidemiologic study is available to assess the exact prevalence for each of these disorders. Previously referred to as mtDNA depletion syndrome 8A (encephalomyopathic type with renal tubulopathy) or Previously referred to as mtDNA depletion syndrome 8B ## Diagnosis Note: While muscle biops...
[ "B Acham-Roschitz, B Plecko, F Lindbichler, R Bittner, CJ Mache, W Sperl, JA Mayr. A novel mutation of the RRM2B gene in an infant with early fatal encephalomyopathy, central hypomyelination, and tubulopathy.. Mol Genet Metab. 2009;98:300-4", "B Bornstein, E Area, KM Flanigan, J Ganesh, P Jayakar, KJ Swoboda, J C...
17/4/2014
24/6/2021
GeneReviews®
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[ "Review", "Clinical Review" ]
rss
rss
[ "Russell-Silver Syndrome", "Russell-Silver Syndrome", "Cyclin-dependent kinase inhibitor 1C", "High mobility group protein HMGI-C", "Insulin-like growth factor 2", "Zinc finger protein PLAG1", "CDKN1C", "H19", "HMGA2", "IGF2", "PLAG1", "Silver-Russell Syndrome" ]
Silver-Russell Syndrome
Howard M Saal, Madeleine D Harbison, Irene Netchine
Summary Silver-Russell Syndrome (SRS) is typically characterized by gestational growth restriction resulting in affected individuals being born small for gestational age, with relative macrocephaly at birth (head circumference ≥1.5 standard deviations [SD] above birth weight and/or length), prominent forehead with fron...
## Diagnosis Consensus clinical diagnostic criteria for Silver-Russell syndrome (SRS) have been published [ SRS Small for gestational age (birth weight and/or length two or more standard deviations [SD] below the mean for gestational age) Postnatal growth failure (length/height two or more SD below the mean for age...
[]
2/11/2002
9/5/2024
9/1/2025
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
rsts
rsts
[ "Broad Thumb-Hallux Syndrome", "Broad Thumbs-Hallux Syndrome", "CREB-binding protein", "Histone acetyltransferase p300", "CREBBP", "EP300", "Rubinstein-Taybi Syndrome" ]
Rubinstein-Taybi Syndrome
Cathy A Stevens
Summary Rubinstein-Taybi syndrome (RSTS) is characterized by distinctive facial features, broad and often angulated thumbs and halluces, short stature, and moderate-to-severe intellectual disability. Characteristic craniofacial features include downslanted palpebral fissures, low-hanging columella, high palate, grimaci...
## Diagnosis Rubinstein-Taybi syndrome (RSTS) Craniofacial appearance (See Downslanted palpebral fissures Convex nasal ridge with low-hanging columella High palate Grimacing smile Talon cusps (an accessory cusp-like structure on the lingual side of the tooth), usually occurring on the maxillary incisors of the...
[]
30/8/2002
9/11/2023
GeneReviews®
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[ "Review", "Clinical Review" ]
rtps
rtps
[ "Rhabdoid Predisposition Syndrome", "RTPS", "Rhabdoid Predisposition Syndrome", "SWI/SNF-related matrix-associated actin-dependent regulator of chromatin subfamily A member 4", "SWI/SNF-related matrix-associated actin-dependent regulator of chromatin subfamily B member 1", "SMARCA4", "SMARCB1", "Rhabd...
Rhabdoid Tumor Predisposition Syndrome
Karolina Nemes, Susanne Bens, Franck Bourdeaut, Pascal Johann, Uwe Kordes, Reiner Siebert, Michael C Frühwald
Summary Rhabdoid tumor predisposition syndrome (RTPS) is characterized by a markedly increased risk for the development of rhabdoid tumors – rare and highly aggressive malignant tumors occurring predominantly in infants and children younger than age three years. Malignant rhabdoid tumors can occur in almost any anatomi...
## Diagnosis Rhabdoid tumor predisposition syndrome (RTPS) Congenital presentation (i.e., prenatal diagnosis or symptoms within the first 28 days of life) Early-onset rhabdoid tumor (age <12 months) Advanced stage of rhabdoid tumor at diagnosis (e.g., >M Synchronous rhabdoid tumors (>1 primary rhabdoid tumor) Fam...
[ "A. Agaimy. SWI/SNF complex-deficient soft tissue neoplasms: a pattern-based approach to diagnosis and differential diagnosis.. Surg Pathol Clin. 2019;12:149-63", "AC Ammerlaan, A Ararou, MP Houben, F Baas, CC Tijssen, JL Teepen, P Wesseling, TJ Hulsebos. Long-term survival and transmission of INI1-mutation via n...
7/12/2017
12/5/2022
GeneReviews®
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[ "Review", "Clinical Review" ]
rts
rts
[ "Anaphase-promoting complex subunit 1", "ATP-dependent DNA helicase Q4", "ANAPC1", "RECQL4", "Rothmund-Thomson Syndrome" ]
Rothmund-Thomson Syndrome
Lisa L Wang, Sharon E Plon
Summary Rothmund-Thomson syndrome (RTS) is characterized by a rash that progresses to poikiloderma; sparse hair, eyelashes, and/or eyebrows; small size; skeletal and dental abnormalities; juvenile cataracts; and an increased risk for cancer, especially osteosarcoma. A variety of benign and malignant hematologic abnorma...
## Diagnosis Rothmund-Thomson syndrome (RTS) Starts in infancy, usually between ages three and six months Erythema on the cheeks and face Spreads to involve the extensor surfaces of the extremities Typically sparing of the trunk and abdomen; possible involvement of the buttocks Gradually develops over a perio...
[ "NF Ajeawung, TM Nguyen, L Lu, TJ Kucharski, J Rousseau, S Molidperee, J Atienza, I Gamache, W Jin, SE Plon, BH Lee, JG Teodoro, LL Wang, PM Campeau. Mutations in ANAPC1, encoding a scaffold subunit of the anaphase promoting complex, cause Rothmund-Thomson syndrome Type 1.. Am J Hum Genet. 2019;105:625-30", "ML B...
6/10/1999
3/1/2019
4/6/2020
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
runx1
runx1
[ "Familial Platelet Disorder / Acute Myeloid Leukemia (FPD/AML)", "RUNX1 Familial Platelet Disorder (FPD)", "Familial Platelet Disorder / Acute Myeloid Leukemia (FPD/AML)", "RUNX1 Familial Platelet Disorder (FPD)", "Runt-related transcription factor 1", "RUNX1", "RUNX1 Familial Platelet Disorder with Ass...
Natalie Deuitch, Elizabeth Broadbridge, Lea Cunningham, Paul Liu
Summary The diagnosis of
## Diagnosis Abnormal bruising or bleeding (90%) Bruising without known trauma Excessive bleeding following surgery or trauma Bleeding from the gums after brushing or flossing teeth or prolonged bleeding following dental cleaning or dental extractions Obstetric and gynecologic manifestations may include menorrha...
[]
4/3/2021
11/1/2024
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
rvcl
rvcl
[ "Cerebroretinal Vasculopathy (CRV)", "Hereditary Endotheliopathy, Retinopathy, Nephropathy, and Stroke (HERNS)", "Hereditary Systemic Angiopathy (HSA)", "Hereditary Vascular Retinopathy (HVR)", "Retinal Vasculopathy with Cerebral Leukodystrophy (RVCL)", "RVCL-S", "RVCL-S", "Retinal Vasculopathy with C...
Retinal Vasculopathy with Cerebral Leukoencephalopathy and Systemic Manifestations
Irene de Boer, Nadine Pelzer, Gisela Terwindt
Summary The diagnosis of RVCL-S is established in a proband with suggestive findings and a heterozygous pathogenic variant in RVCL-S is inherited in an autosomal dominant manner. Most individuals diagnosed with RVCL-S have an affected parent. However, disease onset and severity vary considerably even within the same fa...
## Diagnosis There are no consensus clinical diagnostic criteria for retinal vasculopathy with cerebral leukoencephalopathy and systemic manifestations (RVCL-S). RVCL-S Vascular retinopathy typically manifesting as decreased visual acuity and/or visual field defects Focal and/or global brain dysfunction and brain...
[ "C Carra-Dalliere, X Ayrignac, C Prieto-Morin, P Girard, E Tournier-Lasserve, P. Labauge. TREX1 mutation in leukodystrophy with calcifications and persistent gadolinium-enhancement.. Eur Neurol. 2017;77:113-4", "AC Cohn, K Kotschet, A Veitch, MB Delatycki, MF McCombe. Novel ophthalmological features in hereditary...
19/9/2019
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
rws
rws
[ "Calmodulin", "Calmodulin-3", "Caveolin-3", "Inward rectifier potassium channel 2", "Potassium voltage-gated channel subfamily E member 1", "Potassium voltage-gated channel subfamily E member 2", "Potassium voltage-gated channel subfamily KQT member 1", "Sodium channel protein type 5 subunit alpha", ...
Long QT Syndrome Overview
Alexander J Groffen, Hennie Bikker, Imke Christiaans
Summary The purpose of this overview is to: Briefly describe the Review the Review the Provide an Review Inform
## Clinical Characteristics of Long QT Syndrome Long QT syndrome (LQTS) is characterized by QT prolongation and T wave abnormalities on EKG. LQTS predisposes individuals to a significant risk of life-threatening arrhythmic events, especially in young individuals. Molecular genetic testing identifies a genetic cause i...
[]
20/2/2003
21/3/2024
4/8/2009
GeneReviews®
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[ "Review", "Clinical Review" ]
salih-myo
salih-myo
[ "Titin", "TTN", "Salih Myopathy" ]
Salih Myopathy
Peter Hackman, Marco Savarese, Maria Francesca Di Feo, Bjarne Udd, Mustafa A Salih
Summary Salih myopathy is characterized by muscle weakness (manifesting during the neonatal period or in very early infancy) and delayed motor development; children acquire independent walking between ages 20 months and four years. In the first decade of life, global motor performance is stable or tends to improve. Mod...
## Diagnosis No consensus clinical diagnostic criteria for Salih myopathy have been published. Salih myopathy Muscle weakness manifesting during the neonatal period or in very early infancy Delayed motor milestones but normal cognitive development Muscle weakness of limb-girdle distribution, myopathic face, vari...
[]
12/1/2012
3/4/2025
GeneReviews®
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[ "Review", "Clinical Review" ]
samd9l-ap
samd9l-ap
[ "SAMD9L-ATXPC Syndrome", "SAMD9L-ATXPC Syndrome", "Sterile alpha motif domain-containing protein 9-like", "SAMD9L", "SAMD9L Ataxia-Pancytopenia Syndrome" ]
Wendy H Raskind, Dong-Hui Chen, Thomas Bird
Summary The diagnosis of
## Diagnosis Formal clinical diagnostic criteria for Cerebellar ataxia Variable hematopoietic cytopenias affecting one or more lineages (e.g., anemia, neutropenia, thrombocytopenia) Myeloid leukemia or myelodysplasia with partial or complete monosomy 7 The diagnosis of Note: Identification of a heterozygous Mo...
[]
1/6/2017
4/2/2021
GeneReviews®
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[ "Review", "Clinical Review" ]
sandhoff
sandhoff
[ "Type II GM2 Gangliosidosis", "Type II GM2 Gangliosidosis", "Acute Infantile Sandhoff Disease", "Subacute Juvenile Sandhoff Disease", "Late-Onset Sandhoff Disease", "Beta-hexosaminidase subunit beta", "HEXB", "Sandhoff Disease" ]
Sandhoff Disease
Changrui Xiao, Cynthia Tifft, Camilo Toro
Summary Sandhoff disease comprises a phenotypic continuum encompassing acute infantile, subacute juvenile, and late-onset disease. Although classification into these phenotypes is somewhat arbitrary, it is helpful in understanding the variation observed in the timing of disease onset, presenting manifestations, rate of...
Acute infantile Sandhoff disease Subacute juvenile Sandhoff disease Late-onset Sandhoff disease For synonyms and outdated names see For other genetic causes of these phenotypes see • Acute infantile Sandhoff disease • Subacute juvenile Sandhoff disease • Late-onset Sandhoff disease ## Diagnosis No consensus cl...
[]
14/4/2022
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
satb2-dis
satb2-dis
[ "2q32 Deletion Syndrome", "2q33.1 Microdeletion Syndrome", "Glass Syndrome", "2q32 Deletion Syndrome", "2q33.1 Microdeletion Syndrome", "Glass Syndrome", "DNA-binding protein SATB2", "SATB2", "SATB2-Associated Syndrome" ]
Yuri A Zarate, Katherine Bosanko, Jennifer Fish
Summary The diagnosis of SAS is established in a proband with suggestive findings and identification of one of the following by molecular genetic testing: a heterozygous intragenic SAS is an autosomal dominant disorder. Almost all probands with SAS reported to date have the disorder as the result of a
## Diagnosis No formal clinical diagnostic criteria have been established for SAS Typically moderate-to-profound developmental delay or intellectual disability, including severe speech delay and, in some, absence of speech; however, individuals with milder developmental delay affecting predominantly speech have be...
[]
12/10/2017
20/6/2024
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
saul-wilson
saul-wilson
[ "Microcephalic Osteodysplastic Dysplasia", "Microcephalic Osteodysplastic Dysplasia", "Conserved oligomeric Golgi complex subunit 4", "COG4", "Saul-Wilson Syndrome" ]
Saul-Wilson Syndrome
Carlos Ferreira
Summary Saul-Wilson syndrome (SWS) is a skeletal dysplasia characterized by profound short stature, distinctive craniofacial features, short distal phalanges of fingers and toes, and often clubfoot. Early development (primarily speech and motor) is delayed; cognition is normal. Other findings can include hearing loss (...
## Diagnosis Formal diagnostic criteria for Saul-Wilson syndrome have not been established. Saul-Wilson syndrome Skeletal Profound short stature (typically of prenatal onset) Clubfoot Short distal phalanges of fingers and toes (See Distinctive craniofacial features (See Progeroid facial appearance (more strik...
[ "Y Chinen, T Kaneshi, T Kamiya, K Hata, G Nishimura, T Kaname. Progressive hip joint subluxation in Saul-Wilson syndrome.. Am J Med Genet A. 2015;167A:2834-8", "CR Ferreira, ZJ Xia, A Clément, DA Parry, M Davids, F Taylan, P Sharma, CT Turgeon, B Blanco-Sánchez, BG Ng, CV Logan, LA Wolfe, BD Solomon, MT Cho, G Do...
20/2/2020
GeneReviews®
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[ "Review", "Clinical Review" ]
sca-io
sca-io
[ "IOSCA", "Mitochondrial DNA Depletion Syndrome 7", "IOSCA", "Mitochondrial DNA Depletion Syndrome 7", "Twinkle mtDNA helicase", "TWNK", "Infantile-Onset Spinocerebellar Ataxia" ]
Infantile-Onset Spinocerebellar Ataxia – RETIRED CHAPTER, FOR HISTORICAL REFERENCE ONLY
Tuula Lönnqvist
Summary Infantile-onset spinocerebellar ataxia (IOSCA) is a severe, progressive neurodegenerative disorder characterized by normal development until age one year, followed by onset of ataxia, muscle hypotonia, loss of deep-tendon reflexes, and athetosis. Ophthalmoplegia and sensorineural deafness develop by age seven ...
## Diagnosis Infantile-onset spinocerebellar ataxia (IOSCA) is a clinical spectrum that was originally described in individuals of Finnish descent; however, the phenotype has been expanded by the identification of affected individuals of non-Finnish descent whose features may deviate from the originally described "cla...
[]
27/1/2009
19/4/2018
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sca1
sca1
[ "SCA1", "SCA1", "Ataxin-1", "ATXN1", "Spinocerebellar Ataxia Type 1" ]
Spinocerebellar Ataxia Type 1
Puneet Opal, Tetsuo Ashizawa
Summary Spinocerebellar ataxia type 1 (SCA1) is characterized by progressive cerebellar ataxia, dysarthria, and eventual deterioration of bulbar functions. Early in the disease, affected individuals may have gait disturbance, slurred speech, difficulty with balance, brisk deep tendon reflexes, hypermetric saccades, nys...
## Diagnosis The phenotypic manifestations of spinocerebellar ataxia type 1 (SCA1) are not specific, and no formal clinical diagnostic criteria exist. SCA1 Progressive cerebellar ataxia Dysarthria Eventual deterioration of bulbar functions The diagnosis of SCA1 39-44 CAG repeat alleles must be uninterrup...
[ "T Ashizawa, KP Figueroa, SL Perlman, CM Gomez, GR Wilmot, JD Schmahmann, SH Ying, TA Zesiewicz, HL Paulson, VG Shakkottai, KO Bushara, SH Kuo, MD Geschwind, G Xia, P Mazzoni, JP Krischer, D Cuthbertson, AR Holbert, JH Ferguson, SM Pulst, SH Subramony. Clinical characteristics of patients with spinocerebellar ataxi...
1/10/1998
2/2/2023
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sca10
sca10
[ "SCA10", "SCA10", "Ataxin-10", "ATXN10", "Spinocerebellar Ataxia Type 10" ]
Spinocerebellar Ataxia Type 10
Tohru Matsuura, Tetsuo Ashizawa
Summary Spinocerebellar ataxia type 10 (SCA10) is characterized by slowly progressive cerebellar ataxia that usually starts as poor balance and unsteady gait, followed by upper-limb ataxia, scanning dysarthria, and dysphagia. Abnormal tracking eye movements are common. Recurrent seizures after the onset of gait ataxia ...
## Diagnosis Spinocerebellar ataxia type 10 (SCA10) Slowly progressive cerebellar ataxia starting as poor balance and unsteady gait Scanning dysarthria, dysphagia, and upper-limb ataxia following the gait ataxia Family history consistent with autosomal dominant inheritance and Native American or East Asian ancestry...
[]
23/4/2002
19/9/2019
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sca11
sca11
[ "SCA11", "SCA11", "Tau-tubulin kinase 2", "TTBK2", "Spinocerebellar Ataxia Type 11" ]
Spinocerebellar Ataxia Type 11
Zhongbo Chen, Arina Puzriakova, Henry Houlden
Summary Spinocerebellar ataxia type 11 (SCA11) is characterized by progressive cerebellar ataxia and abnormal eye signs (jerky pursuit, horizontal and vertical nystagmus). Pyramidal features are seen on occasion. Peripheral neuropathy and dystonia are rare. Six families have been reported to date, one each from the UK,...
## Diagnosis Spinocerebellar ataxia type 11 (SCA11) Progressive cerebellar ataxia Abnormal eye signs (jerky pursuit, horizontal and vertical nystagmus) Dysarthria Pyramidal features (mild-to-moderate lower-extremity hyperreflexia; in very rare cases, a positive Babinski sign or other pyramidal features) Swallowin...
[ "I Alesutan, M Sopjani, M Dërmaku-Sopjani, C Munoz, J Voelkl, F Lang. Upregulation of Na-coupled glucose transporter SGLT1 by Tau tubulin kinase 2.. Cell Physiol Biochem. 2012;30:458-65", "P Bauer, G Stevanin, C Beetz, M Synofzik, T Schmitz-Hübsch, U Wüllner, E Berthier, E Ollagnon-Roman, O Riess, S Forlani, E Mu...
22/7/2008
31/10/2019
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sca12
sca12
[ "SCA12", "SCA12", "SCA 12", "Serine/threonine-protein phosphatase 2A 55 kDa regulatory subunit B beta isoform", "PPP2R2B", "Spinocerebellar Ataxia Type 12" ]
Spinocerebellar Ataxia Type 12 – RETIRED CHAPTER, FOR HISTORICAL REFERENCE ONLY
Russell L Margolis, Susan E Holmes, Achal K Srivastava, Mitali Mukherji, KK Sinha
Summary Spinocerebellar ataxia type 12 (SCA12) is characterized by onset of action tremor of the upper extremities in the fourth decade, slowly progressing to include ataxia and other cerebellar and cortical signs. Given the small number of individuals known to have SCA12, it is possible that other clinical manifestat...
## Diagnosis Clinical information on spinocerebellar ataxia type 12 (SCA12) derives from studies of the index pedigree, an American family of German descent [ The diagnosis of SCA12 should be considered in the following: Individuals of Indian descent who: Develop an action tremor of the upper extremities in mid-lif...
[ "S Bahl, K Virdi, U Mittal, MP Sachdeva, AK Kalla, SE Holmes, E O'Hearn, RL Margolis, S Jain, AK Srivastava, M Mukerji. Evidence of a common founder for SCA12 in the Indian population.. Ann Hum Genet 2005;69:528-34", "A Brusco, C Gellera, C Cagnoli, A Saluto, A Castucci, C Michielotto, V Fetoni, C Mariotti, N Mig...
1/10/2004
17/11/2011
12/3/2007
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sca13
sca13
[ "SCA13", "SCA13", "KCNC3 Congenital-Onset Non-Progressive Cerebellar Ataxia", "KCNC3 Childhood-Onset Progressive Cerebellar Ataxia with Delayed Milestones", "KCNC3 Adult-Onset Progressive Cerebellar Ataxia", "Voltage-gated potassium channel KCNC3", "KCNC3", "Spinocerebellar Ataxia Type 13" ]
Spinocerebellar Ataxia Type 13
Michael F Waters
Summary Spinocerebellar ataxia type 13 (SCA13) is a phenotypic spectrum that includes both non-progressive infantile-onset ataxia and progressive childhood-onset and adult-onset cerebellar ataxia. Three phenotypes are seen: Cerebellar hypoplasia with non-progressive infantile-onset limb, truncal, and gait ataxia with m...
Congenital-onset non-progressive cerebellar ataxia Childhood-onset progressive cerebellar ataxia w/delayed milestones Adult-onset progressive cerebellar ataxia For other genetic causes of these phenotypes, see • Congenital-onset non-progressive cerebellar ataxia • Childhood-onset progressive cerebellar ataxia w/de...
[ "K Bürk, DA Sival. Scales for the clinical evaluation of cerebellar disorders.. Handb Clin Neurol. 2018;154:329-39", "K Bürk, A Strzelczyk, PS Reif, KP Figueroa, SM Pulst. Mesial temporal lobe epilepsy in a patient with spinocerebellar ataxia type 13 (SCA13).. Int J Neurosci 2013;123:278-282", "M Coutelier, G C...
9/11/2006
4/6/2020
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sca14
sca14
[ "SCA14", "SCA14", "Protein kinase C gamma type", "PRKCG", "Spinocerebellar Ataxia Type 14" ]
Spinocerebellar Ataxia Type 14
Dong-Hui Chen, Thomas D Bird, Wendy H Raskind
Summary Spinocerebellar ataxia type 14 (SCA14) is characterized by slowly progressive cerebellar ataxia, dysarthria, and nystagmus. Axial myoclonus, cognitive impairment, tremor, and sensory loss may also be observed. Parkinsonian features including rigidity and tremor have been described in some families. Findings see...
## Diagnosis Formal diagnostic criteria for spinocerebellar ataxia type 14 have not been established. Spinocerebellar ataxia type 14 (SCA14) Slowly progressive cerebellar ataxia Myoclonus, dystonia, rigidity, and tremor Sensory loss Dysarthria Nystagmus Cognitive impairment (some individuals) Depression (some ...
[ "N Adachi, T Kobayashi, H Takahashi, T Kawasaki, Y Shirai, T Ueyama, T Matsuda, T Seki, N Sakai, N Saito. Enzymological analysis of mutant protein kinase Cgamma causing spinocerebellar ataxia type 14 and dysfunction in Ca2+ homeostasis.. J Biol Chem. 2008;283:19854-63", "H Asai, M Hirano, K Shimada, T Kiriyama, Y...
28/1/2005
20/2/2020
21/12/2005
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sca15
sca15
[ "SCA15", "SCA15", "SCA 15", "Inositol 1,4,5-trisphosphate receptor type 1", "ITPR1", "Spinocerebellar Ataxia Type 15" ]
Spinocerebellar Ataxia Type 15 – RETIRED CHAPTER, FOR HISTORICAL REFERENCE ONLY
Elsdon Storey
Summary Spinocerebellar ataxia type 15 (SCA15) is characterized by slowly progressive gait and limb ataxia, often in combination with ataxic dysarthria, titubation, upper limb postural tremor, mild hyperreflexia, gaze-evoked nystagmus, and impaired vestibuloocular reflex gain. Onset is between ages seven and 72 years,...
## Diagnosis The diagnosis of spinocerebellar ataxia type 15 (SCA15) should be considered in individuals with the following findings: Very slowly progressive ataxia (e.g., still independently ambulant after 20-30 years of symptoms) No other neurologic signs beyond postural and kinetic tremor (which are common and ma...
[]
30/5/2006
12/6/2014
21/4/2011
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sca17
sca17
[ "Huntington Disease-Like 4", "SCA17", "SCA17", "Huntington Disease-Like 4", "TATA-box-binding protein", "TBP", "Spinocerebellar Ataxia Type 17" ]
Spinocerebellar Ataxia Type 17
Yasuko Toyoshima, Osamu Onodera, Mitsunori Yamada, Shoji Tsuji, Hitoshi Takahashi
Summary Spinocerebellar ataxia type 17 (SCA17) is characterized by ataxia, dementia, and involuntary movements, including chorea and dystonia. Psychiatric symptoms, pyramidal signs, and rigidity are common. The age of onset ranges from three to 55 years. Individuals with full-penetrance alleles develop neurologic and/o...
## Diagnosis Spinocerebellar ataxia type 17 (SCA17) Ataxia Dementia Involuntary movements – e.g., chorea and dystonia (blepharospasm, torticollis, writer's cramp, foot dystonia) Psychiatric symptoms The diagnosis of SCA17 The CAA CAG CAA interruption between (CAG) Molecular genetic testing approaches can includ...
[ "A Alendar, B Euljkovic, D Savic, A Djarmati, M Keckarevic, A Ristic, N Dragasevic, V Kosic, S Romac. Spinocerebellar ataxia type 17 in the Yugoslav population.. Acta Neurol Scand 2004;109:185-7", "A Alibardi, F Squitieri, F Fattapposta, P Missori, F Pierelli, C Trompetto, A Curra. Psychiatric onset and late chor...
29/3/2005
12/9/2019
28/7/2022
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sca2
sca2
[ "SCA2", "SCA 2", "SCA2", "Ataxin-2", "ATXN2", "Spinocerebellar Ataxia Type 2" ]
Spinocerebellar Ataxia Type 2
Stefan M Pulst
Summary Spinocerebellar ataxia type 2 (SCA2) is characterized by progressive cerebellar ataxia, including nystagmus, slow saccadic eye movements, and in some individuals, ophthalmoparesis or parkinsonism. Pyramidal findings are present; deep tendon reflexes are brisk early on and absent later in the course. Age of onse...
## Diagnosis Spinocerebellar ataxia type 2 (SCA2) Slowly progressive ataxia and dysarthria Nystagmus and slow saccadic eye movements Family history consistent with autosomal dominant inheritance The diagnosis of SCA2 Note: Interruption of a CAG expanded allele by a CAA repeat does not mitigate the pathogenicity...
[]
23/10/1998
14/2/2019
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sca20
sca20
[ "SCA20", "SCA20", "Spinocerebellar Ataxia Type 20" ]
Spinocerebellar Ataxia Type 20
Elsdon Storey, RJM Gardner
Summary Spinocerebellar ataxia type 20 (SCA20) is characterized by a slowly progressive ataxia and dysarthria. Approximately two thirds of those affected also display palatal tremor ("myoclonus") and/or abnormal phonation clinically resembling spasmodic adductor dysphonia. Dysarthria, which may be abrupt in onset, prec...
## Diagnosis Spinocerebellar ataxia type 20 (SCA20) Onset with dysarthria (rather than with gait ataxia) that may be abrupt in onset (seen in ~66%) Palatal tremor (in ~66%) Family history consistent with autosomal dominant inheritance Additional findings may include the following: Hypermetric horizontal saccades ...
[ "F Barbieri, MT Pellecchia, E Esposito, E Di Stasio, I Castaldo, F Santorelli, A Perretti, L Santoro, G De Michele. Adult-onset familial laryngeal abductor paralysis, cerebellar ataxia, and pure motor neuropathy.. Neurology 2001;56:1412-4", "JG de Yebenes, A Vazquez, J Rabano, EV de Seijas, DG Urra, MC Obregon, M...
27/2/2007
18/4/2019
6/1/2009
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sca28
sca28
[ "SCA28", "SCA28", "Mitochondrial inner membrane m-AAA protease component AFG3L2", "AFG3L2", "Spinocerebellar Ataxia Type 28" ]
Spinocerebellar Ataxia Type 28
Alessandro Brussino, Alfredo Brusco, Alexandra Durr, Cecilia Mancini
Summary Spinocerebellar ataxia type 28 (SCA28) is characterized by young-adult onset, very slowly progressive gait and limb ataxia resulting in coordination and balance problems, dysarthria, ptosis, nystagmus, and ophthalmoparesis. In most individuals, SCA28 presents as a loss of coordination of lower limbs (unsteadine...
## Diagnosis Spinocerebellar ataxia type 28 (SCA28) Onset generally in young adulthood (but with a wide range: ages 3-76 years) A slowly progressive gait disorder resulting from cerebellar impairment Cerebellar dysarthria Oculomotor abnormalities including ophthalmoparesis, nystagmus and ptosis Hyperreflexia or b...
[]
17/5/2011
22/3/2018
7/2/2013
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sca3
sca3
[ "Machado-Joseph Disease", "SCA3", "Machado-Joseph Disease", "SCA3", "Ataxin-3", "ATXN3", "Spinocerebellar Ataxia Type 3" ]
Spinocerebellar Ataxia Type 3
Henry Paulson, Vikram Shakkottai
Summary Spinocerebellar ataxia type 3 (SCA3), also known as Machado-Joseph disease (MJD), is characterized by progressive cerebellar ataxia and variable findings including pyramidal signs, a dystonic-rigid extrapyramidal syndrome, significant peripheral amyotrophy and generalized areflexia, progressive external ophthal...
## Diagnosis Spinocerebellar ataxia type 3 (SCA3), also known as Machado-Joseph disease (MJD), Pyramidal signs A dystonic-rigid extrapyramidal syndrome Significant peripheral amyotrophy and generalized areflexia Progressive external ophthalmoplegia Action-induced facial and lingual fasciculations; bulging eyes T...
[ "T Ashizawa, G Öz, HL Paulson. Spinocerebellar ataxias: prospects and challenges for therapy development.. Nat Rev Neurol 2018;14:590-605", "P Braga-Neto, JL Pedroso, H Alessi, LA Dutra, AC Felício, T Minett, P Weisman, RF Santos-Galduroz, PH Bertolucci, AA Gabbai, OG Barsottini. Cerebellar cognitive affective sy...
10/10/1998
4/6/2020
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sca36
sca36
[ "Asidan/SCA36", "Costa da Morte Ataxia", "SCA36", "SCA36", "Asidan/SCA36", "Costa da Morte Ataxia", "Nucleolar protein 56", "NOP56", "Spinocerebellar Ataxia Type 36" ]
Spinocerebellar Ataxia Type 36 – RETIRED CHAPTER, FOR HISTORICAL REFERENCE ONLY
Manuel Arias, Beatriz Quintáns, María García-Murias, Maria J Sobrido
Summary Spinocerebellar ataxia type 36 (SCA36) is characterized by a late-onset, slowly progressive cerebellar syndrome typically associated with sensorineural hearing loss. Other common features are muscle atrophy and denervation, especially of the tongue, as well as pyramidal signs, thus overlapping with motor neuro...
## Diagnosis The clinical suspicion of spinocerebellar ataxia type 36 (SCA36) is based on the presence of the following nonspecific findings: Midline cerebellar ataxia of late onset (usually between ages 40 and 60 years) and slow progression Dysarthria and appendicular ataxia generally following the gait imbalance ...
[ "K Abe, Y Ikeda, T Kurata, Y Ohta, Y Manabe, M Okamoto, K Takamatsu, T Ohta, Y Takao, Y Shiro, M Shoji, T Kamiya, H Kobayashi, A. Koizumi. Cognitive and affective impairments of a novel SCA/MND crossroad mutation Asidan.. Eur J Neurol. 2012;19:1070-8", "M Arias, S Arias-Rivas, P Blanco-Arias, D Dapena, F Vázquez,...
7/8/2014
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sca37
sca37
[ "SCA37", "SCA37", "Disabled homolog 1", "DAB1", "Spinocerebellar Ataxia Type 37" ]
Spinocerebellar Ataxia Type 37
Antoni Matilla-Dueñas, Victor Volpini
Summary Spinocerebellar ataxia type 37 (SCA37) is characterized by adult onset, dysarthria, slowly progressive gait and limb ataxia with severe dysmetria in the lower extremities, mild dysmetria in the upper extremities, dysphagia, and abnormal ocular movements (dysmetric vertical saccades, irregular and slow vertical ...
## Diagnosis The phenotypic manifestations of spinocerebellar ataxia type 37 (SCA37) are not specific and no formal diagnostic criteria exist; thus, the diagnosis of SCA37 rests on molecular genetic testing. However, if autosomal dominant inheritance is apparent, or if pure cerebellar ataxia with adult onset, initial ...
[ "M Corral-Juan, C Serrano-Munuera, A Rábano, D Cota-González, A Segarra-Roca, L Ispierto, AT Cano-Orgaz, AD Adarmes, C Méndez-del-Barrio, S Jesús, P Mir, V Volpini, R Alvarez-Ramo, I Sánchez, A Matilla-Dueñas. Clinical, genetic and neuropathological characterisation of spinocerebellar ataxia type 37.. Brain 2018;14...
30/5/2019
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sca38
sca38
[ "SCA38", "Very long chain fatty acid elongase 5", "ELOVL5", "Spinocerebellar Ataxia Type 38" ]
Spinocerebellar Ataxia Type 38
Alfredo Brusco, Eleonora Di Gregorio, Barbara Borroni
Summary Spinocerebellar ataxia type 38 (SCA38) is characterized as a pure cerebellar ataxia with symptoms typically manifesting in the fourth decade of life. The most common presenting features are nystagmus and slowly progressive gait ataxia. As the disease progresses, cerebellar symptoms (limb ataxia, dysarthria, dys...
## Diagnosis Formal clinical diagnostic criteria for spinocerebellar ataxia 38 (SCA38) have not been established. Spinocerebellar ataxia type 38 (SCA38) Slowly progressive gait ataxia with onset in adulthood (3rd-5th decade) Nystagmus in the lateral and vertical gaze Hyposmia Cerebellar atrophy (sometimes r...
[]
11/7/2019
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sca4
sca4
[ "SCA4", "Spinocerebellar Ataxia 4", "SCA4", "Spinocerebellar Ataxia 4", "Zinc finger homeobox protein 3", "ZFHX3", "Spinocerebellar Ataxia Type 4" ]
Spinocerebellar Ataxia Type 4
Andreas Puschmann, Sigurd Dobloug, Joel Wallenius, Klas Wictorin, Sorina Gorcenco
Summary Spinocerebellar ataxia type 4 (SCA4) is a progressive neurologic disease characterized by cerebellar involvement (gait ataxia, balance disturbances, eye movement abnormalities), brain stem involvement (dysarthria, dysphagia), sensory neuropathy, motor neuron involvement (muscle wasting and spasticity), autonomi...
## Diagnosis No consensus diagnostic criteria for spinocerebellar ataxia type 4 (SCA4) have been published. Spinocerebellar ataxia type 4 (SCA4) Age of onset ranges from 12 to 65 years. About 10% of individuals have early onset (i.e., before age 25 years). Although affected individuals may report dizziness when w...
[]
12/12/2024
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sca6
sca6
[ "SCA6", "SCA6", "Voltage-dependent P/Q-type calcium channel subunit alpha-1A", "CACNA1A", "Spinocerebellar Ataxia Type 6" ]
Spinocerebellar Ataxia Type 6
Hannah L Casey, Christopher M Gomez
Summary Spinocerebellar ataxia type 6 (SCA6) is characterized by adult-onset, slowly progressive cerebellar ataxia, dysarthria, and nystagmus. The age of onset ranges from 19 to 73 years; mean age of onset is between 43 and 52 years. Initial symptoms are gait unsteadiness, stumbling, and imbalance (in ~90%) and dysarth...
## Diagnosis Formal diagnostic criteria for spinocerebellar ataxia type 6 (SCA6) have not been established. SCA6 The diagnosis of SCA6 Meiotic expansion of a 19-CAG repeat allele into the known pathogenic range [ Elderly asymptomatic individuals [ An individual with atypical features of SCA6 [ An ataxic indivi...
[]
23/10/1998
21/11/2019
16/6/2008
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sca7
sca7
[ "SCA7", "SCA7", "Ataxin-7", "ATXN7", "Spinocerebellar Ataxia Type 7" ]
Spinocerebellar Ataxia Type 7
Albert R La Spada
Summary Spinocerebellar ataxia type 7 (SCA7) comprises a phenotypic spectrum ranging from adolescent- or adult-onset progressive cerebellar ataxia and cone-rod retinal dystrophy to infantile or early-childhood onset with multiorgan failure, an accelerated course, and early death. Anticipation in this nucleotide repeat ...
## Diagnosis Spinocerebellar ataxia type 7 (SCA7) Progressive incoordination caused by cerebellar ataxia, including dysarthria/dysphagia, dysmetria, and dysdiadochokinesia. Cone-rod retinal dystrophy with the following: Loss of central vision A tritan-axis (blue/yellow) defect on detailed color vision testing M...
[]
27/8/1998
23/7/2020
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sca8
sca8
[ "SCA8", "SCA8", "Ataxin-8", "ATXN8", "ATXN8OS", "Spinocerebellar Ataxia Type 8" ]
Spinocerebellar Ataxia Type 8
John Douglas Cleary, SH Subramony, Laura PW Ranum
Summary SCA8 is a slowly progressive ataxia with onset typically in the third to fifth decade but with a range from before age one year to after age 60 years. Common initial manifestations are scanning dysarthria with a characteristic drawn-out slowness of speech and gait instability. Over the disease course other find...
## Diagnosis Spinocerebellar ataxia type 8 (SCA8) Gait and limb ataxia Scanning dysarthria characterized by a drawn-out slowness of speech Eye movement abnormalities (e.g., nystagmus, abnormal pursuit and abnormal saccades) Often "extracerebellar signs" including: Upper motor neuron findings (e.g., brisk tendon r...
[]
27/11/2001
22/4/2021
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
scad
scad
[ "SCADD", "SCAD Deficiency", "SCAD Deficiency", "SCADD", "Short-chain specific acyl-CoA dehydrogenase, mitochondrial", "ACADS", "Short-Chain Acyl-CoA Dehydrogenase Deficiency" ]
Short-Chain Acyl-CoA Dehydrogenase Deficiency
Lynne Wolfe, Reena Jethva, Devin Oglesbee, Jerry Vockley
Summary Most infants with short-chain acyl-CoA dehydrogenase deficiency (SCADD) identified through newborn screening programs have remained well, and asymptomatic relatives who meet diagnostic criteria are reported. Thus, SCADD is now viewed as a biochemical phenotype rather than a disease. A broad range of clinical fi...
## Diagnosis Short-chain acyl-CoA dehydrogenase deficiency (SCADD) has been defined as the presence of: Increased butyrylcarnitine (C4) concentrations in plasma and/or increased ethylmalonic acid (EMA) concentrations in urine under non-stressed conditions (on at least two occasions) AND Biallelic Most infants with...
[]
22/9/2011
9/8/2018
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
scan1
scan1
[ "SCAN1, TDP1-Related Spinocerebellar Ataxia with Axonal Neuropathy", "SCAN1", "TDP1-Related Spinocerebellar Ataxia with Axonal Neuropathy", "Tyrosyl-DNA phosphodiesterase 1", "TDP1", "Spinocerebellar Ataxia with Axonal Neuropathy Type 1" ]
Spinocerebellar Ataxia with Axonal Neuropathy Type 1
Mustafa AM Salih, Hiroshi Takashima, Cornelius F Boerkoel
Summary Spinocerebellar ataxia with axonal neuropathy type 1 (SCAN1) is characterized by late-childhood-onset slowly progressive cerebellar ataxia and distal sensorimotor axonal neuropathy. Gaze nystagmus and dysarthria usually develop after the onset of ataxic gait. As the disease advances, pain and touch sensation in...
## Diagnosis No consensus clinical diagnostic criteria for spinocerebellar ataxia with axonal neuropathy type 1 (SCAN1) have been published. SCAN1 is suspected in individuals with the following clinical findings, electrophysiologic studies, laboratory findings, brain imaging, and family history [ Slowly progressiv...
[]
22/10/2007
30/6/2022
20/12/2012
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
schaaf-yang
schaaf-yang
[ "Chitayat-Hall Syndrome", "Chitayat-Hall Syndrome", "MAGE-like protein 2", "MAGEL2", "Schaaf-Yang Syndrome" ]
Schaaf-Yang Syndrome
Christian P Schaaf, Felix Marbach
Summary Schaaf-Yang syndrome (SYS) is a rare neurodevelopmental disorder that shares multiple clinical features with the genetically related The diagnosis of Schaaf-Yang syndrome is established in a proband by identification of a heterozygous pathogenic variant in the paternally derived Schaaf-Yang syndrome is inherite...
## Diagnosis Formal clinical diagnostic criteria for Schaaf-Yang syndrome (SYS) have not been established. SYS Generalized hypotonia of infancy Respiratory distress in infancy Infant feeding difficulties with failure to thrive Hyperphagia with subsequent obesity in childhood or adolescence Mild-to-profound dev...
[]
11/2/2021
4/11/2021
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
schinzel-giedion
schinzel-giedion
[ "Classic Schinzel-Giedion Syndrome", "Atypical Schinzel-Giedion Syndrome", "SET-binding protein", "SETBP1", "Schinzel-Giedion Syndrome" ]
Schinzel-Giedion Syndrome
Jessica Duis, Bregje WM van Bon
Summary Classic Schinzel-Giedion syndrome (SGS), an ultra-rare multisystem disorder caused by gain-of-function pathogenic variants in a To date, more than 50 individuals have been reported with molecularly confirmed classic SGS. Atypical SGS, reported in five individuals to date, is caused by pathogenic The diagnosis o...
Pathogenic variants in Spectrum of Phenotypes Associated with Broad spectrum of clinical features of variable severity that partially overlap w/classic SGS Atypical SGS is milder than classic SGS. " Note: " See • Broad spectrum of clinical features of variable severity that partially overlap w/classic SGS • A...
[]
7/3/2024
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
schmid-mcd
schmid-mcd
[ "Metaphyseal Chondrodysplasia Type Schmid (MCDS)", "Metaphyseal Dysplasia Schmid (MCS), COL10A1-Related", "Metaphyseal Chondrodysplasia Type Schmid (MCDS)", "Metaphyseal Dysplasia Schmid (MCS), COL10A1-Related", "Collagen alpha-1(X) chain", "COL10A1", "Schmid Metaphyseal Chondrodysplasia" ]
Schmid Metaphyseal Chondrodysplasia
Christopher Mark Richmond, Ravi Savarirayan
Summary Schmid metaphyseal chondrodysplasia (SMCD) is characterized by progressive short stature that develops by age two years. The clinical and radiographic features are usually not present at birth, but manifest in early childhood with short limbs, genu varum, and waddling gait. Facial features and head size are nor...
## Diagnosis No formal diagnostic criteria for Schmid metaphyseal chondrodysplasia (SMCD) have been established. SMCD Short-limbed short stature by age two years (in >60%) Genu varum (bowed legs) (>60%) Waddling gait (>80%) Lumbar lordosis by age three to five years Normal craniofacies and absence of extraskel...
[]
21/10/2019
9/5/2024
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
schwann
schwann
[ "Congenital Cutaneous Neurilemmomatosis", "Leucine-zipper-like transcriptional regulator 1", "SWI/SNF-related matrix-associated actin-dependent regulator of chromatin subfamily B member 1", "LZTR1", "SMARCB1", "Schwannomatosis" ]
Radhika Dhamija, Scott Plotkin, Alicia Gomes, Dusica Babovic-Vuksanovic
Summary The diagnosis of
## Diagnosis Consensus diagnostic criteria for Two or more non-intradermal tumors suggestive of schwannomas Absence of bilateral vestibular schwannomas A family history of schwannomatosis consistent with autosomal dominant inheritance (e.g., affected males and females in multiple generations). Absence of a known fa...
[]
8/3/2018
27/7/2023
25/4/2024
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
scn3a-ndd
scn3a-ndd
[ "Sodium channel protein type 3 subunit alpha", "SCN3A", "SCN3A-Related Neurodevelopmental Disorder" ]
Katherine L Helbig, Ethan M Goldberg
Summary The diagnosis of
## Diagnosis Clinical diagnostic criteria for Intractable seizures beginning in the first year of life, particularly in the first month of life (median age 2 weeks) Developmental delay or intellectual disability, often in the severe-to-profound range in those with early-onset developmental and epileptic encephalop...
[]
3/6/2021
1/5/2025
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
scn8a-ee
scn8a-ee
[ "SCN8A-Related Developmental and Epileptic Encephalopathy (DEE)", "SCN8A-Related Mild-to-Moderate Developmental and Epileptic Encephalopathy (mild/modDEE)", "SCN8A-Related Self-Limited Familial Infantile Epilepsy (SeLFIE)", "SCN8A-Related Neurodevelopmental Disorder with Generalized Epilepsy (NDDwGE)", "SCN...
Michael F Hammer, Maya Xia, John M Schreiber
Summary The diagnosis of
For other genetic causes of these phenotypes, see ## Diagnosis No consensus clinical diagnostic criteria for Childhood-onset seizures: seizure onset variable, ranges from the first few months to the first few years of life Development of multiple seizure types, including focal, multifocal, or generalized seizur...
[]
25/8/2016
6/4/2023
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
scs
scs
[ "Acrocephalosyndactyly Type III", "Acrocephalosyndactyly Type III", "Twist-related protein 1", "TWIST1", "Saethre-Chotzen Syndrome" ]
Saethre-Chotzen Syndrome
Emily R Gallagher, Chootima Ratisoontorn, Michael L Cunningham
Summary Classic Saethre-Chotzen syndrome (SCS) is characterized by coronal synostosis (unilateral or bilateral), facial asymmetry (particularly in individuals with unicoronal synostosis), strabismus, ptosis, and characteristic appearance of the ear (small pinna with a prominent superior and/or inferior crus). Syndactyl...
## Diagnosis Saethre-Chotzen syndrome (SCS) Craniosynostosis (premature fusion of one or more sutures of the calvarium) The coronal suture is the most commonly affected, although any or all sutures can be affected. Craniosynostosis often presents with an abnormal skull shape (e.g., brachycephaly [short, broad skull...
[ "P Bourgeois, AL Bolcato-Bellemin, JM Danse, A Bloch-Zupan, K Yoshiba, C Stoetzel, F Perrin-Schmitt. The variable expressivity and incomplete penetrance of the twist-null heterozygous mouse phenotype resemble those of human Saethre-Chotzen syndrome.. Hum Mol Genet 1998;7:945-57", "J Cai, BK Goodman, AS Patel, JB ...
16/5/2003
24/1/2019
14/6/2012
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sd-thes
sd-thes
[ "Phenotypic Diarrhea of Infancy", "Syndromic Diarrhea/Tricho-Hepato-Enteric Syndrome (SD/THE)", "THES", "THES", "Syndromic Diarrhea/Tricho-Hepato-Enteric Syndrome (SD/THE)", "Phenotypic Diarrhea of Infancy", "Tricho-Hepato-Enteric Syndrome", "Superkiller complex protein 2", "Superkiller complex prot...
Trichohepatoenteric Syndrome
Alexandre Fabre, Patrice Bourgeois, Charlène Chaix, Karine Bertaux, Olivier Goulet, Catherine Badens
Summary Trichohepatoenteric syndrome (THES), generally considered to be a neonatal enteropathy, is characterized by intractable diarrhea (seen in almost all affected children), woolly hair (seen in all), intrauterine growth restriction, facial dysmorphism, and short stature. Additional findings include poorly character...
## Diagnosis To date, no diagnostic algorithm for trichohepatoenteric syndrome (THES) has been published. THES * The association of neonatal intractable diarrhea and IUGR suggests the diagnosis of THES. The diagnosis of THES Note: (1) Per ACMG/AMP variant interpretation guidelines, the terms "pathogenic variant" a...
[]
11/1/2018
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sds
sds
[ "Shwachman-Bodian-Diamond Syndrome", "Shwachman-Bodian-Diamond Syndrome", "DnaJ homolog subfamily C member 21", "Elongation factor-like GTPase 1", "Ribosome maturation protein SBDS", "Signal recognition particle subunit SRP54", "DNAJC21", "EFL1", "SBDS", "SRP54", "Shwachman-Diamond Syndrome" ]
Shwachman-Diamond Syndrome
Adam Nelson, Kasiani Myers
Summary Shwachman-Diamond syndrome (SDS) is characterized by exocrine pancreatic dysfunction with malabsorption, malnutrition, and growth failure; hematologic abnormalities with single- or multilineage cytopenias and susceptibility to myelodysplastic syndrome (MDS) and acute myelogenous leukemia (AML); and bone abnorma...
## Diagnosis No consensus clinical diagnostic criteria for Shwachman-Diamond syndrome (SDS) have been published. SDS Low serum concentrations of the pancreatic enzymes trypsinogen and/or isoamylase for age. Note: Measurement of trypsinogen concentration should be used in children age 3 years [ Low levels of fecal...
[]
17/7/2008
19/9/2024
GeneReviews®
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[ "Review", "Clinical Review" ]
sedt
sedt
[ "TRAPPC2-Related X-Linked Spondyloepiphyseal Dysplasia Tarda", "TRAPPC2-Related X-Linked Spondyloepiphyseal Dysplasia Tarda", "Trafficking protein particle complex subunit 2", "TRAPPC2", "X-Linked Spondyloepiphyseal Dysplasia Tarda" ]
X-Linked Spondyloepiphyseal Dysplasia Tarda
George E Tiller
Summary In adults, X-linked spondyloepiphyseal dysplasia tarda (X-linked SEDT) is characterized by disproportionately short stature with short trunk and arm span significantly greater than height. At birth, affected males are normal in length and have normal body proportions. Affected males exhibit linear growth defici...
## Diagnosis No consensus clinical diagnostic criteria for X-linked spondyloepiphyseal dysplasia tarda have been published. X-linked spondyloepiphyseal dysplasia tarda (X-linked SEDT) Disproportionate short stature in adolescence or adulthood and a relatively short trunk and barrel-shaped chest. Upper- to lower-body...
[ "J Fiedler, M Le Merrer, G Mortier, S Heuertz, L Faivre, RE Brenner. X-linked spondyloepiphyseal dysplasia tarda: Novel and recurrent mutations in 13 European families.. Hum Mutat 2004;24:103", "J Gécz, MA Hillman, AK Gedeon, TC Cox, E Baker, JC Mulley. Gene structure and expression study of the SEDL gene for spo...
1/11/2001
5/11/2020
6/4/2023
GeneReviews®
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[ "Review", "Clinical Review" ]
serine-def
serine-def
[ "Serine Biosynthesis Disorders", "Serine Synthesis Disorders", "Serine Biosynthesis Disorders", "Serine Synthesis Disorders", "Neu-Laxova Syndrome", "D-3-phosphoglycerate dehydrogenase", "Phosphoserine aminotransferase", "Phosphoserine phosphatase", "PHGDH", "PSAT1", "PSPH", "Serine Deficiency...
Serine Deficiency Disorders
Saskia N van der Crabben, Tom J de Koning
Summary Serine deficiency disorders include a spectrum of disease ranging from lethal prenatal-onset Neu-Laxova syndrome to serine deficiency with infantile, juvenile, or adult onset. Neu-Laxova syndrome is characterized by severe intrauterine growth deficiency, microcephaly, congenital bilateral cataracts, characteris...
Serine Deficiency Disorders: Phenotypic Spectrum Infantile-onset phenotype: severe neurodevelopmental disorder w/epilepsy & microcephaly Juvenile-onset phenotype: developmental & behavioral issues & epilepsy Adult-onset phenotype: progressive axonal neuropathy, variable ataxia &/or cognitive impairment For other ge...
[]
22/6/2023
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
setbp1-hd
setbp1-hd
[ "SETBP1 Disorder", "SETBP1 Disorder", "SET-binding protein", "SETBP1", "SETBP1 Haploinsufficiency Disorder" ]
Angela Morgan, Siddharth Srivastava, Jessica Duis, Bregje van Bon
Summary The diagnosis of
Pathogenic variants in Spectrum of Phenotypes Associated with ## Diagnosis No consensus clinical diagnostic criteria for Motor developmental delay (in 97%) Developmental delay / mild-to-severe intellectual disability Learning difficulties Speech and language disorder (including childhood apraxia of speech) ...
[]
18/11/2021
9/5/2024
GeneReviews®
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[ "Review", "Clinical Review" ]
setd1b-ndd
setd1b-ndd
[ "Histone-lysine N-methyltransferase SETD1B", "SETD1B", "SETD1B-Related Neurodevelopmental Disorder" ]
Alexandra Roston, William Gibson
Summary The diagnosis of
## Diagnosis Developmental delay (especially speech and language delay) Intellectual disability (ID) Seizures that are frequently refractory to treatment Autism spectrum disorder or autism-like behaviors Other behavioral concerns (hyperactivity, aggression, anxiety, sleep disorders) The diagnosis of Note: (1) Pe...
[]
29/9/2022
27/2/2025
GeneReviews®
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[ "Review", "Clinical Review" ]
setd2-ndd
setd2-ndd
[ "SETD2 Neurodevelopmental Disorder with or without Macrocephaly/Overgrowth", "SETD2 Neurodevelopmental Disorder with Multiple Congenital Anomalies", "Histone-lysine N-methyltransferase SETD2", "SETD2", "SETD2 Neurodevelopmental Disorders" ]
John Pappas, Rachel Rabin
Summary The diagnosis of a
For other genetic causes of these phenotypes, see ## Diagnosis No consensus clinical diagnostic criteria for Congenital heart defects Urogenital anomalies Ophthalmologic findings including Low anterior hairline Biparietal narrowing Flat face with maxillary hypoplasia Arched eyebrows Widely spaced eyes Shor...
[]
30/12/2021
22/9/2022
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sft
sft
[ "Bifunctional UDP-N-acetylglucosamine 2-epimerase/N-acetylmannosamine kinase", "GNE", "Sialuria" ]
Sialuria – RETIRED CHAPTER, FOR HISTORICAL REFERENCE ONLY
Jules G Leroy
Summary Sialuria is characterized by variable and transient signs and symptoms, especially in infancy. These include slightly flat and coarse facies, prolonged neonatal jaundice, equivocal or mild hepatomegaly, microcytic anemia, frequent upper respiratory infections, and episodes of gastroenteritis, dehydration, and ...
## Diagnosis The diagnosis of sialuria may be suspected in infants or young children with the following: Mild facial coarsening Hypotonia Equivocal developmental delay Frequent upper respiratory infections Note: The likelihood of sialuria is increased after exclusion of more prevalent disorders that share laborat...
[ "Z Argov, I Eisenberg, G Grabov-Nardini, M Sadeh, I Wirguin, D Soffer, S Mitrani-Rosenbaum. Hereditary inclusion body myopathy: the Middle Eastern genetic cluster.. Neurology. 2003;60:1519-23", "Z Argov, S Mitrani-Rosenbaum. The hereditary inclusion body myopathy enigma and its future therapy.. Neurotherapeutics....
14/1/2004
18/10/2012
27/2/2007
GeneReviews®
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[ "Review", "Clinical Review" ]
sgbs
sgbs
[ "Glypican-3", "GPC3", "Simpson-Golabi-Behmel Syndrome Type 1" ]
Simpson-Golabi-Behmel Syndrome Type 1
Alex F Nisbet, Evan R Hathaway, Jennifer M Kalish
Summary Simpson-Golabi-Behmel syndrome type 1 (SGBS1) is characterized by pre- and postnatal macrosomia; distinctive craniofacial features (including macrocephaly, coarse facial features, macrostomia, macroglossia, and palate abnormalities); and, commonly, mild-to-severe intellectual disability with or without structur...
## Diagnosis For the purposes of this Consensus clinical diagnostic criteria for Simpson-Golabi-Behmel syndrome type 1 (SGBS1) have not been established. SGBS1 Widely spaced eyes, epicanthal folds, and downslanted palpebral fissures Redundant, furrowed skin over the glabella Wide nasal bridge and antevert...
[]
19/12/2006
7/12/2023
14/11/2024
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sgpl1
sgpl1
[ "SGPL1 Deficiency", "Steroid-Resistant Nephrotic Syndrome Type 14", "SGPL1 Deficiency", "Steroid-Resistant Nephrotic Syndrome Type 14", "Sphingosine-1-phosphate lyase 1", "SGPL1", "Sphingosine Phosphate Lyase Insufficiency Syndrome" ]
Sphingosine Phosphate Lyase Insufficiency Syndrome
Kathryn Nicole Weaver, Bonnie Sullivan, Friedhelm Hildebrandt, Jonathan Strober, Megan Cooper, Rathi Prasad, Julie Saba
Summary Sphingosine phosphate lyase insufficiency syndrome (SPLIS) is characterized by varying combinations of steroid-resistant nephrotic syndrome (ranging from nonimmune fetal hydrops to adolescent onset), primary adrenal insufficiency (with or without mineralocorticoid deficiency), testicular insufficiency, hypothyr...
## Diagnosis Sphingosine phosphate lyase insufficiency syndrome (SPLIS) Primary adrenal insufficiency (low cortisol with normal or high ACTH). Typically, glucocorticoid deficiency; some individuals also have mineralocorticoid deficiency. Testicular insufficiency (increased gonadotropins, poor response to LH stim...
[]
15/10/2020
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sgs
sgs
[ "Ski oncogene", "SKI", "Shprintzen-Goldberg Syndrome" ]
Shprintzen-Goldberg Syndrome
Marie T Greally
Summary Shprintzen-Goldberg syndrome (SGS) is characterized by: delayed motor and cognitive milestones and mild-to-moderate intellectual disability; craniosynostosis of the coronal, sagittal, or lambdoid sutures; distinctive craniofacial features; and musculoskeletal findings including olichostenomelia, arachnodactyly,...
## Diagnosis Formal diagnostic criteria for Shprintzen-Goldberg syndrome (SGS) have not been established. SGS Dolichocephaly with or without scaphocephaly Tall or prominent forehead Hypertelorism Downslanting palpebral fissures Ocular proptosis Malar flattening High narrow palate with prominent palatine ridg...
[ "LC Adès, K Sullivan, A Biggin, EA Haan, M Brett, KJ Holman, J Dixon, S Robertson, AD Holmes, J Rogers, B Bennetts. FBN1, TGFBR1, and the Marfan-craniosynostosis/mental retardation disorders revisited.. Am J Med Genet A. 2006;140:1047-58", "PY Au, HE Racher, JM Graham, N Kramer, RB Lowry, JS Parboosingh, AM Innes...
13/1/2006
9/4/2020
GeneReviews®
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[ "Review", "Clinical Review" ]
shashi-pena
shashi-pena
[ "SHAPNS", "SHAPNS", "Putative Polycomb group protein ASXL2", "ASXL2", "Shashi-Pena Syndrome" ]
Shashi-Pena Syndrome
Julie M Porter, Loren DM Pena, Rebecca C Spillmann, Amanda Johnson, Vandana Shashi
Summary Shashi-Pena syndrome is characterized by distinctive facial features accompanied by variable further clinical findings. Facial features may include glabellar nevus simplex, widely spaced and prominent/proptotic eyes with epicanthal folds and ptosis, arched eyebrows, broad nasal tip, and low-set/posteriorly rota...
## Diagnosis No consensus clinical diagnostic criteria for Shashi-Pena syndrome have been published. Shashi-Pena syndrome Distinctive facial features (most recognizable in infancy and becoming less discernible in older individuals) (see Nevus simplex in the glabellar region of the forehead Widely spaced and prom...
[]
7/11/2024
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
short
short
[ "Phosphatidylinositol 3-kinase regulatory subunit alpha", "PIK3R1", "SHORT Syndrome" ]
SHORT Syndrome
A Micheil Innes, David A Dyment
Summary SHORT syndrome is a mnemonic for The diagnosis of SHORT syndrome is established in a proband with compatible clinical features (with emphasis on the facial gestalt) and a heterozygous pathogenic variant in SHORT syndrome is inherited in an autosomal dominant manner. The proportion of individuals with SHORT synd...
## Diagnosis The designation SHORT syndrome was coined by SHORT syndrome Intrauterine growth restriction Short stature Partial lipodystrophy Characteristic facial gestalt (see Axenfeld-Rieger anomaly or related anterior chamber ocular anomalies Delayed dentition Insulin resistance / diabetes mellitus No forma...
[]
15/5/2014
4/6/2020
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sickle
sickle
[ "Sickle Cell Disease Due to Hb S/S", "Sickle-Hemoglobin C Disease (Hb S/C)", "Sickle Beta-Thalassemia", "Hemoglobin subunit beta", "HBB", "Sickle Cell Disease" ]
Sickle Cell Disease
MA Bender, Katie Carlberg
Summary Sickle cell disease (SCD) is characterized by intermittent vaso-occlusive events and chronic hemolytic anemia. Vaso-occlusive events result in tissue ischemia leading to acute and chronic pain as well as organ damage that can affect any organ system, including the bones, spleen, liver, brain, lungs, kidneys, an...
Homozygous p.Glu6Val (Hb S/S) Sickle cell disease due to Hb S/S Compound heterozygosity for p.Glu6Val (HbS) and a second Sickle-hemoglobin C disease (Hb S/C) Sickle beta-thalassemia (Hb S/β HbS and another pathogenic beta globin chain variant (e.g., Hb S/D, Hb S/O For synonyms and outdated names see • Homozygous...
[]
15/9/2003
17/11/2022
13/2/2025
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
sider-anemia
sider-anemia
[ "ATP-binding cassette sub-family B member 7, mitochondrial", "ABCB7", "X-Linked Sideroblastic Anemia and Ataxia" ]
X-Linked Sideroblastic Anemia and Ataxia – RETIRED CHAPTER, FOR HISTORICAL REFERENCE ONLY
Soumeya Bekri, Marc D'Hooghe, Pieter Vermeersch
Summary X-linked sideroblastic anemia and ataxia (XLSA/A) is characterized by moderate anemia and early-onset spinocerebellar syndrome in males, manifest primarily as delayed walking, ataxia evident in early childhood, dysmetria, and dysdiadochokinesis. When present the intention tremor is mild and the dysarthria is m...
## Diagnosis Ataxia and incoordination. Note: Upper motor neuron (UMN) signs (i.e., brisk deep tendon reflexes, unsustained ankle clonus, and equivocal or extensor plantar responses) in the legs (present in some males) Mild asymptomatic hypochromic, microcytic anemia Hematocrit ranges from 26% to 35%. Mean corpusc...
[ "R Allikmets, WH Raskind, A Hutchinson, ND Schueck, M Dean, DM Koeller. Mutation of a putative mitochondrial iron transporter gene (ABC7) in X-linked sideroblastic anemia and ataxia (XLSA/A).. Hum Mol Genet. 1999;8:743-9", "S Bekri, G Kispal, H Lange, E Fitzsimons, J Tolmie, R Lill, DF Bishop. Human ABC7 transpor...
1/3/2006
3/4/2014
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
siod
siod
[ "SMARCAL1-Related Immuno-osseous Dysplasia (Schimke Type)", "SWI/SNF-related matrix-associated actin-dependent regulator of chromatin subfamily A-like protein 1", "SMARCAL1", "Schimke Immunoosseous Dysplasia" ]
Schimke Immunoosseous Dysplasia
Elizabeth Lippner, Thomas Lücke, Carlos Salgado, Cornelius Boerkoel, David B Lewis
Summary Schimke immunoosseous dysplasia (SIOD) is characterized by spondyloepiphyseal dysplasia (SED) resulting in short stature, nephropathy, and T cell deficiency. Radiographic manifestations of SED include ovoid and mildly flattened vertebral bodies, small ilia with shallow dysplastic acetabular fossae, and small de...
## Diagnosis No consensus clinical diagnostic criteria for Schimke immunoosseous dysplasia (SIOD) have been published. SIOD Ovoid and mildly flattened vertebral bodies; endplate irregularities, wedged vertebrae, and narrowed vertebral spaces have been reported. Small ilia with hypoplastic basilar portions and...
[ "CE Bansbach, R Bétous, CA Lovejoy, GG Glick, D Cortez. The annealing helicase SMARCAL1 maintains genome integrity at stalled replication forks.. Genes Dev. 2009;23:2405-14", "CE Bansbach, CF Boerkoel, D Cortez. SMARCAL1 and replication stress: An explanation for SIOD?. Nucleus. 2010;1:245-8", "A Baradaran-Hera...
1/10/2002
14/4/2022
30/3/2023
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
slc12a5-e
slc12a5-e
[ "Early-Infantile Epileptic Encephalopathy 34 (EIEE34)", "SLC12A5-EIMFS", "Early-Infantile Epileptic Encephalopathy 34 (EIEE34)", "SLC12A5-EIMFS", "Solute carrier family 12 member 5", "SLC12A5", "SLC12A5-Related Epilepsy of Infancy with Migrating Focal Seizures" ]
Amy McTague, Manju A Kurian
Summary The diagnosis of
## Diagnosis Since 2010 a description of the characteristic symptoms and findings of epilepsy of infancy with migrating focal seizures (EIMFS) has been included in the classification of epilepsy syndromes by the International League Against Epilepsy. The diagnosis of Seizure onset before age six months Development...
[ "C Barba, F Darra, R Cusmai, E Procopio, C Dionisi Vici, L Keldermans, S Vuillaumier-Barrot, DJ Lefeber, R Guerrini, E Parrini, A Ashikov, A Bordugo, G Cantalupo, G Casara, BD Bernardina, M Falchi, L Ferri, D Martinelli, A Morrone, V Race, A Rosati, E Souche. Congenital disorders of glycosylation presenting as epil...
14/2/2019
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
slc19a1-ft-def
slc19a1-ft-def
[ "Reduced folate transporter", "SLC19A1", "SLC19A1-Related Folate Transport Deficiency" ]
I David Goldman
Summary The diagnosis of
## Diagnosis No consensus clinical diagnostic criteria for Birth weight and head circumference that are in the lower ranges of the typical growth chart for age and sex Poor postnatal growth Developmental delay, including gross motor (gait), speech/language, and cognitive skills Seizures Recurrent infections, pa...
[]
14/8/2025
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
slc25a24-fps
slc25a24-fps
[ "Hutchinson-Gilford Progeria-Like Syndrome", "Fontaine-Farriaux Syndrome", "Gorlin-Chaudhry-Moss Syndrome (GCMS)", "Mitochondrial adenyl nucleotide antiporter SLC25A24", "SLC25A24", "SLC25A24 Fontaine Progeroid Syndrome" ]
Danita Velasco, Ann Haskins Olney, Lois Starr
Summary The diagnosis of
## Diagnosis No consensus clinical diagnostic criteria for Pre- and postnatal growth failure Sagging, thin, and translucent skin with decreased subcutaneous fat, contributing to a progeroid appearance, most pronounced in infancy Distinctive craniofacial features ( Cranial underossification with large anterior fo...
[ "N Adolphs, M Klein, EJ Haberl, L Graul-Neumann, H Menneking, B Hoffmeister. Necrotizing soft tissue infection of the scalp after fronto-facial advancement by internal distraction in a 7-year old girl with Gorlin-Chaudhry-Moss syndrome--a case report.. J Craniomaxillofac Surg. 2011;39:554-61", "SR Braddock, HH Ar...
9/6/2022
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
slc39a14-def
slc39a14-def
[ "Hypermanganesemia with Dystonia 2 (HMNDYT2)", "SLC39A14-Related Early-Onset Dystonia-Parkinsonism", "SLC39A14-Related Early-Onset Dystonia-Parkinsonism", "Hypermanganesemia with Dystonia 2 (HMNDYT2)", "Metal cation symporter ZIP14", "SLC39A14", "SLC39A14 Deficiency" ]
SLC39A14 Deficiency
Karin Tuschl, Allison Gregory, Esther Meyer, Peter T Clayton, Susan J Hayflick, Philippa B Mills, Manju A Kurian
Summary SLC39A14 deficiency is typically characterized by evidence of delay or loss of motor developmental milestones (e.g., delayed walking, gait disturbance) between ages six months and three years. Early in the disease course, children show axial hypotonia followed by dystonia, spasticity, dysarthria, bulbar dysfunc...
## Diagnosis SLC39A14 deficiency Delay in acquisition of developmental motor milestones or loss of developmental motor milestones Progressive pharmaco-resistant dystonia Parkinsonism signs (tremor, bradykinesia, hypomimia) Bulbar dysfunction Dysarthria Note: One individual with onset of dystonia during the secon...
[ "KA Alhasan, W Alshuaibi, MH Hamad, S Salim, DZ Jamjoom, AH Alhashim, MA AlGhamdi, AY Kentab, FA Bashiri. Hypermanganesemia with dystonia type 2: a potentially treatable neurodegenerative disorder: a case series in a tertiary university hospital.. Children (Basel) 2022;9:1335", "S Anazi, S Maddirevula, E Faqeih, ...
25/5/2017
8/12/2022
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
slc39a8-cdg
slc39a8-cdg
[ "CDG-IIn", "Congenital Disorder of Glycosylation Type IIn (CDG2N)", "SLC39A8 Deficiency", "CDG-IIn", "Congenital Disorder of Glycosylation Type IIn (CDG2N)", "SLC39A8 Deficiency", "Metal cation symporter ZIP8", "SLC39A8", "SLC39A8-CDG" ]
SLC39A8-CDG
Julien H Park
Summary SLC39A8-CDG is characterized by mild-to-profound developmental delay, intellectual disability, hypotonia, feeding difficulties with poor weight gain and growth deficiency, dystonia, spasticity, epilepsy, ophthalmologic manifestations including cortical blindness and strabismus, and sensorineural hearing impairm...
## Diagnosis SLC39A8-CDG Mild-to-profound developmental delay and/or intellectual disability Generalized hypotonia of infancy Feeding difficulties with poor weight gain and growth deficiency Movement disorder with marked dystonia Spasticity Epilepsy, especially severe infantile epileptic spasms not responding ...
[]
6/4/2023
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
slc6a1-ndd
slc6a1-ndd
[ "SLC6A1-Related Disorder", "SLC6A1 Deficiency Disorder", "SLC6A1-Related Disorder", "SLC6A1 Deficiency Disorder", "Sodium- and chloride-dependent GABA transporter 1", "SLC6A1", "SLC6A1-Related Neurodevelopmental Disorder" ]
Kimberly Goodspeed, Scott Demarest, Katrine Johannesen, Jingqiong Kang, Dennis Lal, Katie Angione
Summary The diagnosis of
## Diagnosis Mild-to-severe developmental delay (DD) and/or intellectual disability (ID) Generalized hypotonia of infancy Epilepsy including absence or atypical absence seizures, epilepsy with myoclonic-atonic seizures, generalized tonic-clonic seizures Movement disorders such as tremor, stereotypies, and ataxia ...
[ "JM Bain, LG Snyder, KL Helbig, DD Cooper, WK Chung, K Goodspeed. Consistency of parent-report SLC6A1 data in Simons Searchlight with provider-based publications.. J Neurodev Disord. 2022;14:40", "GL Carvill, JM McMahon, A Schneider, M Zemel, CT Myers, J Saykally, J Nguyen, A Robbiano, F Zara, N Specchio, O Mecar...
9/2/2023
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]
slc6a3-dtds
slc6a3-dtds
[ "DAT Deficiency", "DAT Deficiency", "Classic Early-Onset Dopamine Transporter Deficiency Syndrome (DTDS)", "Atypical Later-Onset Dopamine Transporter Deficiency Syndrome (DTDS)", "Sodium-dependent dopamine transporter", "SLC6A3", "SLC6A3-Related Dopamine Transporter Deficiency Syndrome" ]
Robert VV Spaull, Manju A Kurian
Summary The diagnosis of In most individuals reported to date, Once the
Classic early-onset dopamine transporter deficiency syndrome (DTDS) Atypical later-onset DTDS • Classic early-onset dopamine transporter deficiency syndrome (DTDS) • Atypical later-onset DTDS ## Diagnosis Classic early-onset and atypical later-onset Onset usually within the first six months of life Early nonsp...
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27/7/2017
28/9/2023
GeneReviews®
https://www.ncbi.nlm.nih.gov/books/NBK1116/
[ "Review", "Clinical Review" ]