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Conclusions
OSA
Within the limitations of this study, we conclude that there is no significant difference in the effects of an MAD allowing limited vertical opening and an MAD allowing free vertical opening on respiratory parameters and upper airway dimensions in patients with mild to moderate OSA.
PMC10160211
Clinical relevance
OSA
MADs allowing limited vertical opening and allowing free vertical opening have similar effects on respiratory parameters and upper airway dimensions in patients with mild to moderate OSA. Trial registration: ClinicalTrials.gov Identifier: NCT02724865.
PMC10160211
Keywords
PMC10160211
Introduction
obstructions of the upper airway, arousals, OSA
OBSTRUCTIVE SLEEP APNEA
Obstructive sleep apnea (OSA) is characterized by recurrent obstructions of the upper airway, often resulting in oxygen desaturations and arousals from sleep [Continuous positive airway pressure (CPAP) is the most efficacious therapy for OSA [There is a large variety of commercially available customized MADs. Over the years, the customized MAD has evolved from the “mono-bloc” type of appliance, which consists of a single piece giving the mandible a fixed position, towards the current “bi-block titratable” type, which consists of two separate pieces that are dynamically interconnected allowing different degree of lateral and/or vertical movements. Understanding the treatment efficacy and working mechanism of MADs with different design features has an ongoing interest in the clinic [Bi-block titratable MADs may differ in the freedom of mandibular vertical opening. There are two types of bi-block titratable MADs that are commonly used in the treatment of OSA: the Herbst appliance (MAD-H) and the SomnoDent appliance (MAD-S). They predominantly differ in the freedom of vertical opening: the MAD-H only allows limited vertical opening, while the MAD-S allows free vertical opening of the mandible during sleep. A retrospective study suggested no significant difference between MAD-H and MAD-S in changing the AHI [The working mechanism of MAD is related to the improvement of the upper airway dimensions through mandibular protrusion [We hypothesized that MAD-H (allowing limited vertical opening) would lead to better improvement in respiratory parameters and upper airway dimensions compared to MAD-S (allowing free vertical opening), especially in supine position. This hypothesis was based primarily on the notion that gravity may promote more mandibular opening in the MAD-S group in the supine position, which subsequently results in less improvement of the upper airway CSAmin [
PMC10160211
Material and methods
PMC10160211
Overview
This study was a multi-center RCT with a parallel design, in which the effects of two types of titratable MADs on respiratory parameters and upper airway dimensions were compared. The allocation sequence was automatically generated through an electronic data capture system (Castor EDC), using random block size of either 4, 6, or 8 with an allocation ratio of 1:1. The Medical Research Ethics Committee of the Academic Medical Center Amsterdam (AMC) approved this study (#: NL44085.018.13). The study was registered at clinicaltrials.gov (ClinicalTrials.gov identifier: NCT02724865). Written informed consent was obtained from all participants.
PMC10160211
Participants
OSA symptoms, OSA, apnea, daytime sleepiness, snoring
Eligible patients, diagnosed with OSA at four sleep centers in the Netherlands (Onze Lieve Vrouwe Gasthuis Ziekenhuis (OLVG), Nederlands Slaap Instituut, Medisch Centrum Jan van Goyen, and AMC), were referred to the department of Oral and Maxillofacial Surgery of AMC to participate in the present study. The inclusion criteria were: 1. ≥ 18 years old; 2. ability to speak, read, and write Dutch; 3. ability to follow-up; 4. ability to use a computer with internet connection for online questionnaires; 5. diagnosis with symptomatic mild or moderate OSA (5 ≤ apnea–hypopnea index (AHI) ˂ 30 events/hour) with at least two OSA symptoms (e.g., snoring, fragmented sleep, witnessed apneas, and/or excessive daytime sleepiness determined by Epworth Sleepiness Scale (ESS) [
PMC10160211
MADs
The two types of MAD compared in this study were MAD-H (Herbst appliance; 4Dental labs, Amsterdam, the Netherlands) and MAD-S (SomnoDent appliance; SomnoDent Flex, SomnoMed, Sydney, Australia). The MAD-H consisted of two splints connected to each other with adjustable iron-bars for titration. The vertical opening was limited by these bars (Fig. Mandibular advancement devices (MADs). (The standardized titration [
PMC10160211
MAD side-effects and compliance
temporomandibular complaints
The self-reported side effects were recorded at each clinical visit, including the following: 1. sensitive teeth in the morning; 2. painful jaw muscles; 3. painful temporomandibular complaints; and 4. changed occlusion in the morning. The compliance information was collected by a telephone survey, which consisted of four questions: 1. number of hours of MAD use per night; 2. number of hours of total sleep per night; 3. number of days of MAD use per week; and 4. the overall level of satisfaction of the MAD usage. Level of satisfaction was based on a visual analogue scale of 0 to 100, where 0 was unsatisfied, and 100 was very satisfied. The compliance data were expressed as percentage of hours of MAD use per total sleep time, and as percentage of days of MAD use per week.
PMC10160211
Polysomnography (PSG) recordings
Every patient underwent a baseline PSG recording and a 3-month follow-up PSG recording with MAD in situ at one of the afore-mentioned sleep centers. A digital PSG system (Embla ASleep and respiration were analyzed following the criteria of the American Academy of Sleep Medicine (AASM) Task Force [
PMC10160211
Cone beam computed tomography (CBCT)
epiglottis, overbite
All patients underwent two CBCT scans (NewTom 5G, QR systems, Italy) at the department of Oral Radiology of ACTA: a baseline scan and a follow-up scan with the MAD in situ (in the same protrusion position as during the follow-up PSG recording). The exposure settings were 110 kV, 4 mA, 0.3 mm voxel size, 3.6-s exposure time (pulsed radiation), and 18–36-s scanning time, depending on the size of the patient [Using Amira® software (v4.1, Visage Imaging Inc., Carlsbad, CA, USA), upper airway segmentation was performed by applying the superior and inferior boundaries of the upper airway. The superior boundary of the upper airway was the palatal plane, and the inferior boundary was the horizontal plane across the base of the epiglottis (parallel to the palatal plane) (Fig. Measurements of the upper airway dimensions using cone beam computed tomography (CBCT) imaging. (All of the upper airway variables were measured by an experienced examiner. To test the intra-rater reliability of the assessment of the upper airway, 10 CBCT scans were randomly selected and re-measured after a 1-month interval of the original measurements.Based on baseline and follow-up CBCT images, the vertical opening with the MAD in situ was determined in two steps in 3Diagnosys ® software (v5.3.1, 3diemme, Cantu, Italy). Firstly, the overbite was measured based on baseline CBCT images (Fig. Measurements of mandibular vertical opening using cone beam computed tomography (CBCT) imaging. (
PMC10160211
Sample size
According to the guidelines of Cohen, an effect size d, which is defined as a standardized difference in the means of an outcome variable between two groups, of 0.20 is small, one of 0.5 is medium, and one of 0.8 is large [
PMC10160211
Statistical analysis
SECONDARY
Normality of continuous data was tested by Shapiro–Wilk test. Independent The intra-rater reliability for the upper airway variables was assessed using a two-way mixed, absolute agreement, single measures intraclass correlation coefficient (ICC). Reliability was defined as poor (ICC < 0.5), moderate (ICC = 0.5–0.75), good (ICC = 0.75–0.9), or excellent (ICC > 0.9) [Two-way analyses of variance (ANOVA) were used to compare the mean differences of all outcome variables separately for within-subjects factor (i.e., baseline without MAD versus follow-up with MAD in situ), for between-subjects factor (i.e., the mean of the outcome variables of MAD-H versus MAD-S), and to assess the interaction effect between the two factors (i.e., treatment effect between baseline and follow-up of MAD-H versus MAD-S) on the outcome variables. Any significantly different baseline characteristics between both MAD groups were controlled as covariates in the assessment of the interaction effect [A per-protocol (PP) analysis included patients who completed the entire treatment and was performed for all primary and secondary outcomes variables. An intention-to-treat (ITT) analysis included all patients who underwent randomization and was performed only for the primary outcome variables. In ITT analysis, the missing data were imputed by a multiple imputation procedure (five imputed datasets) in SPSS software (SPSS version 20, Chicago, IL, USA). A post hoc power analysis was conducted for the primary outcome variables using the software G*power (version 3.1.9, Franz Faul, Universität Kiel, Germany).
PMC10160211
Results
PMC10160211
Flowchart of participants
OSA
OBSTRUCTIVE SLEEP APNEA
The flow-chart of 49 patients with OSA who were recruited for this study is shown in Fig. Flowchart of the patients in the study. OSA: obstructive sleep apnea; CBCT: cone beam computed tomography; PSG: Polysomnography; n: sample size; MAD: mandibular advancement device; MAD-H: Herbst appliance; MAD-S: SomnoDent appliance
PMC10160211
Reliability of the upper airway assessment
SECONDARY
The intra-rater reliability for the upper airway assessment was excellent, with ICC = 0.96 for the CSAmin and ICC = 0.90 to 0.94 for the secondary outcome variables.
PMC10160211
Discussion
OSA
The aim of this RCT was to compare the effects of MAD-H (allowing limited vertical opening) and MAD-S (allowing free vertical opening) on respiratory parameters and upper airway dimensions in patients with mild to moderate OSA. The results showed that despite differences in the freedom of mandibular vertical opening, there was no significant difference between MAD-H and MAD-S in improving the respiratory parameters and upper airway dimensions.
PMC10160211
MAD-H vs MAD-S
teeth and painful jaw muscles
MINOR
In the present study, there was no significant difference between the MAD-H and MAD-S in affecting the AHI-supine, as well as AHI and AHI-non-supine. According to the post hoc power analysis, the effect size f of the AHI-supine was 0.29 in the PP analysis and was 0.10 in the ITT analysis, which is qualified as small to medium (an effect size f = 0.10 is small, one = 0.25 is medium, and one = 0.40 is large [No significant difference was found between both MADs in changing the upper airway dimensions. With the same protrusion position and vertical opening, the same effect of MAD-H and MAD-S was applied to the tongue and mandible in the awake state, and thus no difference of upper airway changes could be found between both groups. Our results are similar to the outcomes of two previous studies based on cephalometric images [The self-reported side effects were not significantly different between both MAD groups. However, sensitive teeth and painful jaw muscles were 3–4 times more frequent in the MAD-H group compared to MAD-S group, which might be due to the different design feature. However, it seems that the side effects of MAD in most cases were of a minor nature and probably improve in the longer term [
PMC10160211
Overall MAD effects
enlargement
ENLARGEMENT
For respiratory parameters, there was significant decrease in the AHI and AHI-non-supine but no significant decrease in the AHI-supine with MAD in situ in the total group, which indicated that the MADs used in this study were less effective in reducing the AHI in supine position. As MADs used in this study allow more or less freedom of vertical opening, gravity would favor mouth opening and weaken the beneficial effects of mandibular protrusion in supine position for both MADs. A study of Milano et al. [The treatment response rate in the present study (48%) is in line with other studies reporting MAD response rate between 43 and 77% [The CSAmin increased significantly with MAD in situ in the total group. Further, the increase of CSAmin was mainly due to the enlargement in the lateral dimension, which is similar to previous studies [
PMC10160211
Limitations
OSA
The present study has several limitations. Firstly, the information of the patients who were initially screened for this study was missing. With these data missing, we cannot be certain that our sample represents the total population of OSA patients that are being seen in the clinics in Amsterdam. Although the dropout rate (36%) seemed high in our study, the dropout rate was comparable to our previous studies [
PMC10160211
Strengths
The standardized titration protocol used for both types of MAD is one of the major strengths of the present study. To the best of our knowledge, when comparing different MADs, few previous studies have reported a standardized titration protocol. In the present study, a detailed standardized titration protocol was applied, and no significant difference was found in finial protrusion position of both MAD groups, which enables an unbiased comparison. The standardized stepwise titration protocol for MAD, developed by one of the co-authors of this paper (GA), has been proved to have good efficacy, good tolerance, and good adherence [Inserting an MAD intra-orally induces a certain amount of mandibular vertical opening. A study of Mayoral et al. [
PMC10160211
Acknowledgements
The authors gratefully acknowledge Dr. Naichuan Su, Department of Oral Public Health, Academic Centre for Dentistry Amsterdam (ACTA), for his assistance with the statistical analyses of this study; and the staff of the OLVG, Netherlands Slaap Instituut, Medisch Centrum Jan van Goyen and AMC for their assistance with this work.
PMC10160211
Funding
Xiaoxin Shi has received a scholarship from the China Scholarship Council.
PMC10160211
Data availability
The datasets generated and/or analyzed during the current study are available from the corresponding author on reasonable request.
PMC10160211
Declarations
PMC10160211
Ethics approval
This study was performed in line with the principles of the Declaration of Helsinki. Approval was granted by the Medical Research Ethics Committee of the Academic Medical Center Amsterdam (AMC) (#: NL44085.018.13).
PMC10160211
Informed consent
Informed consent was obtained from all individual participants included in the study.
PMC10160211
Conflict of interest
FRANK
Xiaoxin Shi declares that she has no conflicts of interest. Frank Lobbezoo is a member of the Academic Advisory Boards for Grind Care and Oral Function of Sunstar Suisse S.A. and receives research grants from Sunstar Suisse S.A., SomnoMed, Vivisol, Health Holland, and Airway Management. Hui Chen declares that she has no conflicts of interest. Boudewijn R.A.M. Rosenmöller declares that he has no conflicts of interest. Erwin Berkhout declares that he has no conflicts of interest. Jan de Lange declares that he has no conflicts of interest. Ghizlane Aarab is a member of the Academic Advisory Board for Oral Function of Sunstar Suisse S.A. and receives research grants from Sunstar Suisse S.A., SomnoMed, Vivisol, and Health Holland.
PMC10160211
References
PMC10160211
Introduction
Plakoutsis, injuries, Motor behaviors, ’
Evidence has shown that imagining a complex action, like backward-walking, helps improve the execution of the gesture. Despite this, studies in sport psychology have provided heterogeneous results on the use of motor imagery (MI) to improve performance. We aimed to fill this gap by analyzing how sport experience influences backward-walking MI processes in a sample of young women (Open access funding provided by Università degli Studi di Napoli Federico II within the CRUI-CARE Agreement.Motor imagery (MI) describes the mental execution of a movement, motor act, or action without consciously moving or activating muscles (Decety, MI's features enable it to be of value in accelerating procedural learning and recovering motor skills after injuries (Plakoutsis et al., MI can be divided into two categories based on the perspective used during imagination: Athletes’ expertise should also be considered when evaluating the MI process. It has been proven that professional athletes have better imaginative abilities compared to novices since they regularly use MI to improve their gestures (Diotaiuti et al., Lastly, the complexity of the gesture to be imagined represents another crucial issue. Motor behaviors consist of gestures that can be increasingly complex, such as movements, motor acts, and actions (Mandolesi et al., The potential benefits of backward MI training and the growing research on this topic, driven, however, by different experimental protocols and methods as well as conflicting findings (Grealy & Shearer, To this aim, we tested a sample of young women divided into Active and Sedentary in a mental chronometric task in which they had first to imagine and then perform the same motor behavior: forward and backward-walking. Isochrony Efficiency (IE) index was calculated after the task by measuring the difference between imagination and execution times in both conditions (forward and backward).Moreover, we tested how the ability to vividly imagine a motor behavior within various perspectives (visual internal, visual external, and kinesthetic) differently affected IE for active and sedentary individuals. Given the lack of heterogeneous data on the predominant type of perspective used in MI (Morone et al.,
PMC10858124
Materials and methods
PMC10858124
Participants
psychiatric
MOTOR DISORDERS
We recruited 41 women from the “Federico II” University of Naples, divided into two experimental groups: active group (Inclusion criteria were normal or corrected-to-normal vision and right-handedness. Alternatively, exclusion criteria comprised the current or past presence of psychopathology, psychiatric, neurological, or motor disorders, or other medical illness. The participants were voluntarily enrolled after written informed consent was obtained. The study was approved by the Local Ethics Committee of the “Federico II” University of Naples (protocol number: 11/2020) and carried out in accordance with the Declaration of Helsinki.
PMC10858124
Imagery task
A mental chronometry task was administered to participants to assess their motor imagery abilities. There were two randomly assigned tasks in the procedure: a forward-walking task (FW) and a backward-walking task (BW). In both tasks (each consisting of one trial), participants had first to imagine (forward and backward-walking) and then walk (forwards and backward). The corridor used in the present study was situated within a university building and it was familiar to all participants. According to the procedure used by Grealy and Shearer (At the beginning of the procedure, participants were asked to observe a target (a black leather chair) placed 30 m away from their position. We opted for a distance of 30 m, exceeding that used in other protocols (such as Decety et al., For both imagination and execution tasks, each participant was positioned on an “X” placed behind a starting line.Imagination and execution times were measured using a commercial digital stopwatch (Faviye stopwatch XL 0–13). Participants received instructions for using the stopwatch before the imagination task: they had to close their eyes, put their finger on the start button of the stopwatch, and press it as soon as they started imagining. For the execution phase, time was recorded by the experimenter. The times recorded have been measured in seconds. A few days before the test, participants were informed about MI, its use and benefits, and received minimum information about its execution. Each task was evaluated using an Isochrony Efficiency index (IE = difference between imagination and execution time). The bigger IE index accounted for the worst MI efficiency.
PMC10858124
Self-report assessment
The Vividness of Movement Imagery Questionnaire (VMIQ-2) (Roberts et al.,
PMC10858124
Data analysis
One-way ANOVA was used to separately analyzed differences between groups (Active, Sedentary) in the forward and the backward imagery task. To investigate whether imagination perspectives (internal, external, kinesthetic) affect the performance of active and sedentary participants differently, we performed a generalized linear model (GLM) (Dunn & Smyth, All analyses and graph illustrations were conducted through JASP software 0.17.2.1.Due to our relatively small sample size, we conducted a post-hoc power analysis, which revealed a power level (1- β) of 0.34.
PMC10858124
Discussion
Fournier
SECONDARY, FOURNIER
The present study aimed to gain insight into complex action imagery processes, such as backward-walking, as they can be useful in developing training protocols based on MI. Specifically, our main hypothesis concerns the influence of physical activity and sport performance on MI abilities. We tested a sample of young women, divided into Active and Sedentary, in a mental chronometric task in which they had first to imagine and then execute a simple (forward-walking) and a complex (backward-walking) motor behavior. Isochrony Efficiency index (IE), calculated as the difference between imagination and execution times in both conditions (forward and backward), was used.Our findings showed that active individuals, who are experienced in sports, performed better in backward-walking imagination than Sedentary. Active individuals showed a superior IE index (i.e., a smaller difference between imagination and execution times) (Table These results can be discussed in relation to the physical and mental features of both tasks (forward and backward), considering that task difficulty influences temporal accuracy (Calmels & Fournier, The better performance of active individuals in comparison to Sedentary in the backward imagination task can be explained by the fact that physically more active people (like our Active group) physically train different fine motor skills more often, so they have a better, more detailed, and more varied representation of different fine motor skills. The present finding is in line with previous results that evidenced how motor experience is an essential factor for the accuracy of imagery timing (Calmels & Fournier, Our secondary hypothesis regarded the ability to vividly imagine motor behaviors within various perspectives (visual internal, visual external, and kinesthetic) in sedentary and active individuals and how the use of different perspectives was influenced by physical activity and sport experience. We administered the Vividness of Movement Imagery Questionnaire (VMIQ-2) to active and sedentary participants and used VMIQ-2 scales as predictors for MIE in a generalized linear model (GLM).GLM outputs (Table These findings showed that the ability to imagine is indeed influenced by the perspective used. Nevertheless, this seems to apply mostly to sedentary individuals rather than active ones. The association between EVI perspective and poor imagery abilities in FW may be explained by the fact that FW is a simple motor pattern. Therefore, reproducing it from the perspective typically used for learning new movements was inefficient (Montuori et al., The use of MI in sport psychology remains of particular interest, despite all methodological issues discussed so far. MI could be essential for creating training protocols suitable for beginners or more experienced athletes (Fusco et al.,
PMC10858124
Limitations and future directions
Given its preliminary nature, this study presents some limitations. First, our sample consisted entirely of young adult women. Since gender differences significantly influence the processes of mental imagery, testing young adult men is essential to gain stronger insights and extend these results to the general population. Another limit concerns the sample size and the type of action imagined backward. We have chosen a complex imagining task, such as walking backward. Since the distance to be covered appears to be an essential factor in the equivalence of motor imagery timing (Papaxanthis et al.,
PMC10858124
Author contributions
Conceptualization: LM, FL; methodology: NP, FL; investigation: LM, NP, FL; data curation: NP, FL; Validation: LM, NP, FL; formal analysis: NP, FL; visualization: NP; writing—original draft: LM, NP; writing—review & editing: LM, FL.
PMC10858124
Funding
Open access funding provided by Università degli Studi di Napoli Federico II within the CRUI-CARE Agreement. This research was supported by funding from the Department of Humanities, University of Naples Federico II (Fondi ricerca dipartimentale 2023) to L.M. This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.
PMC10858124
Data availability
The data are available from the corresponding author upon request
PMC10858124
Declarations
PMC10858124
Conflict of interest
The authors declare that there are no conflicts of interest and that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.
PMC10858124
Ethical approval
The study was approved by Ethical Committee of Psychological Research of the Department of Humanities of the University of Naples Federico II (n. prot. 11/2020) and was conducted in accordance with the Declaration of Helsinki. Written informed consent was obtained from all individual participants included in the study.
PMC10858124
References
PMC10858124
Introduction
death, HIV treatment initiation, viral suppression
The HIV Prevention Trials Network (HPTN) 074 study demonstrated a positive effect of an integrated systems navigation and psychosocial counseling intervention on HIV treatment initiation, viral suppression, medication assisted treatment (MAT) enrollment, and risk of death among people who inject drugs (PWID). In this sub-study, we analyzed the incidence, causes, and predictors of death among HIV-infected and uninfected participants.
PMC10173611
Methods
The HPTN 074 randomized clinical trial was conducted in Indonesia, Ukraine, and Vietnam. HIV-infected PWID with unsuppressed viral load (indexes) were recruited together with at least one of their HIV-negative injection partners. Indexes were randomized in a 1:3 ratio to the intervention or standard of care.
PMC10173611
Results
depression, deaths, death
The trial enrolled 502 index and 806 partner participants. Overall, 13% (66/502) of indexes and 3% (19/806) of partners died during follow-up (crude mortality rates 10.4 [95% CI 8.1–13.3] and 2.1 [1.3–3.3], respectively). These mortality rates were for indexes nearly 30 times and for partners 6 times higher than expected in a population of the same country, age, and gender (standardized mortality ratios 30.7 [23.7–39.0] and 5.8 [3.5–9.1], respectively). HIV-related causes, including a recent CD4 < 200 cells/μL, accounted for 50% of deaths among indexes. Among partners, medical conditions were the most common cause of death (47%). In the multivariable Cox model, the mortality among indexes was associated with sex (male versus female aHR = 4.2 [1.5–17.9]), CD4 count (≥ 200 versus < 200 cells/μL aHR = 0.3 [0.2–0.5]), depression (moderate-to-severe versus no/mild aHR = 2.6 [1.2–5.0]) and study arm (intervention versus control aHR = 0.4 [0.2–0.9]). Among partners, the study arm of the index remained the only significant predictor (intervention versus control aHR = 0.2 [0.0–0.9]) while controlling for the effect of MAT (never versus ever receiving MAT aHR = 2.4 [0.9–7.4]).
PMC10173611
Conclusions
death
The results confirm that both HIV-infected and uninfected PWID remain at a starkly elevated risk of death compared to general population. Mortality related to HIV and other causes can be significantly reduced by scaling-up ART and MAT. Access to these life-saving treatments can be effectively improved by flexible integrated interventions, such as the one developed and tested in HPTN 074.
PMC10173611
Supplementary Information
The online version contains supplementary material available at 10.1186/s12879-023-08201-3.
PMC10173611
Keywords
PMC10173611
Introduction
death, opioid dependence, overdose, AIDS-related, initiation and retention
People who inject drugs (PWID) are at higher risk of death compared to their non-drug using peers. The all-cause mortality rate in a global systematic review of cohort studies among PWID was 2.9 per 100 person-years (PY), which is nearly 15 times higher than expected in an age-adjusted population [Widespread scale-up of antiretroviral therapy (ART) has led to a dramatic decline in all-cause mortality among people living with HIV (PLWH) worldwide. Unfortunately, these benefits have not been fully extended to PWID due to multiple structural, psychosocial, and other barriers, including lack of treatment for opioid dependence [Effective treatment of opioid dependence leads to reduction in drug use and increased engagement and adherence to other life-saving treatments, radically reducing overdose and AIDS-related mortality in short- and long-term [Numerous interventions have been developed and tested to improve initiation and retention in HIV care and OAT for PWID. Many were shown to have the desired effect in observational or experimental designs [The HIV Prevention Trials Network (HPTN) 074 study was designed to evaluate the feasibility and efficacy of an integrated, scalable intervention aimed at improving ART uptake and viral suppression [In this sub-study, we analyze the incidence of death in a variety of demographic and clinical subgroups, summarize the causes of death in this cohort, and explore predictors of mortality among PWID enrolled in the HPTN 074 trial.
PMC10173611
Methods
PMC10173611
Population and setting
HIV INFECTION, RECRUITMENT
The study was conducted at three sites (Jakarta, Indonesia; Kyiv, Ukraine; and Thai Nguyen, Vietnam). The sites were selected based on estimated high HIV incidence and prevalence among PWID. Participants were recruited in a city hospital (Jakarta), two district health centers (Thai Nguyen), and a community-based harm reduction program (Kyiv). The main recruitment methods were community outreach and peer referral. Study enrollment occurred between April of 2015 and June of 2016 and follow-up in the main phase continued until June 2017.All participants were required to be between 18–45 years of age (the maximum allowed age increased to 60 years after 8 months into the study), active injectors (defined as have injected at least 12 times in the past 3 months), planning to stay in the study area for one year, and able and willing to provide informed consent. Additional criteria for index participants included confirmed HIV infection, HIV viral load ≥ 1,000 copies/mL, CD4 count > 50 cells/μL, and ability to recruit at least one HIV-negative injection partner. Partners had to have HIV-negative test results at screening and have a confirmed relationship with the index (verified by matching the physical description).
PMC10173611
Data collection
death
The study procedures and data collection methods for the main outcomes are presented in detail in the main study report [All instances of death were systematically investigated by the local study teams. After becoming aware of a possible death case, study personnel contacted relatives, peers or medical staff to confirm the death and ascertain the date and the cause of death. All case report forms were independently assessed by two independent physicians to verify and categorize the cause; discrepancies were resolved by a third physician.
PMC10173611
Statistical analysis
depression, deaths, death
DISEASE, DELETION
Crude mortality rates per 100 person years (CMR) were calculated based upon person-time between study enrollment and either the date of death or last follow-up visit. For time-varying covariates, the amount of person time in each category was determined based on the covariate value at the end of each follow-up interval. Exact confidence intervals for crude mortality rates were calculated using the Poisson distribution.Standardized mortality ratios (SMR) were calculated as the observed number of deaths in each subgroup divided by the number of deaths that would be expected during the registered person-time. The mean annual numbers of expected deaths were calculated using the data from the Global Burden of Disease Study, and computed separately for males and females, each 5-year age category, and each country [We used Cox proportional hazard models to analyze predictors of mortality. Unadjusted models included each variable of interest on its own, and adjusted models included the variable of interest in addition to age, gender, site, and study arm. Multivariable models included at the first step all variables significant in the adjusted cox models with a conservative cutoff point of 0.1 and then stepwise deletion was used to remove non-significant variables from the final models. All covariates collected after baseline (e.g. ART use, CD4, viral load, OAT, depression, substance use, stigma) were analyzed as time-varying covariates in the model. Missing data in the longitudinal sequence was imputed from either the most recent visit with non-missing data (for categorical variables), or the mean value between the previous and following visits (continuous variables). Analyses were performed using SAS Version 9.4.Due to substantial differences in mortality rates and causes between HIV-positive indexes and HIV-negative partners, the two groups were analyzed separately.This trial was registered on ClinicalTrials.gov, NCT02935296, on 17/10/2016. The study protocol is available at
PMC10173611
Results
The trial enrolled 502 index (194 [39%] in Vietnam, 187 [37%] in Ukraine, and 121 [24%]) in Indonesia) and 806 partner participants (305 [38%] in Vietnam, 318 [39%] in Ukraine, and 183 [23%]) in Indonesia). A majority were men (87%, 1143/1308); most women (77%, 127/165) enrolled at the Ukrainian site. The median age at enrollment was 34 years (IQR: 30, 38), with no significant difference by study group. Participants in Indonesia were slightly younger compared to Ukraine and Vietnam (median age 32, 34, 35, respectively). A majority reported opioid use in the past three months before baseline (99%, 1299/1308). The median lifetime duration of drug injecting was 13 years (range 0–37) and the median number of injecting days per month was 25 (range 0–31). Detailed baseline characteristics of the study cohort are presented in a previous report [Median follow-up time was 1.2 years (IQR: 0.9–1.5); 3.8% of indexes (9/502) and 6.6% of partners (29/806) were lost to follow-up, and their survival status could not be verified.
PMC10173611
Discussion
deaths, overdose
In this analysis of the randomized clinical trial conducted at three sites in Indonesia, Vietnam, and Ukraine, we found that PWID, regardless of their HIV status, have significantly higher mortality rates than expected in a population of the same age and gender. The majority of deaths among HIV-infected indexes were related to HIV. The number of deaths due to overdose was low in both participant groups. The intervention, primarily focused on ART initiation and adherence, had a significant effect on mortality for both HIV-infected indexes and their HIV-uninfected partners. Mortality rates differed by country, consistently with the uptake of the intervention [
PMC10173611
Mortality rates
AIDS, TB, death, overdose, HIV infected, deaths
AIDS, LIVER DISEASES, TUBERCULOSIS
The observed crude mortality rates were consistent with the recent systematic review of cohort studies, reporting the pooled CMR of 2.71/100PY among people who inject opioids [In our study, the risk of death among PWID living with HIV who were not treated effectively was thirty times higher than expected in a general population adjusted by age and gender, which is twice as high as the pooled SMR in the systematic review. The HIV-uninfected PWID had almost six times higher risk of death compared to the general population, primarily due to acute and chronic health conditions and injuries.There were marked differences in mortality rates between the sites. Among male indexes, mortality in Indonesia was about twice as high as in Vietnam and Ukraine. In contrast, the male partner mortality was highest in Ukraine, with almost two times difference with Indonesia, and three times with Vietnam. These findings correlate with the differences in uptake of ART and OAT across three sites in our study [We were not able identify published sources on PWID mortality for Indonesia and Ukraine. We can, however, compare the mortality rates we observed in Vietnam to two intervention trials conducted in Thai Nguyen and a cohort study in Hai Phong provinces. In the first study, conducted in 2005–2007 among male PWID, 23% of whom were HIV infected [If we attribute mortality with unconfirmed cause in participants who met immunologic criteria for AIDS (CD4 < 200 cells/μL) to HIV, the HIV-related causes would be responsible for more than half of all deaths among index participants. The majority of confirmed HIV-related deaths were due to tuberculosis. This confirms the fact that PWID continue to face significant barriers related to life-saving HIV and TB treatment engagement and adherence in all three countries. Other health-related causes such as lung or liver diseases were the second most common cause of death in HIV-infected, and the leading cause of death in HIV-negative participants in our study.Surprisingly, we observed a low rate of death due to overdose, which accounted for almost 1/3 of mortality among opioid injectors in other studies [
PMC10173611
The effect of the intervention
deaths, death
In our study, we demonstrated a clear and strong effect of the intervention on death outcomes. Among indexes, mortality in the intervention arm compared to the SoC was decreased by more than half. As evident from the multivariable models, the effect of the intervention appears to be mediated by improved immune status, characterized by the CD4 count. Although improved CD4 counts can be plausibly caused only by initiation of ART and resulting viral suppression, these variables did not seem to have an independent effect on mortality in our analysis. Of note, HIV care guidelines since 2015 permitted ART initiation for PWID regardless of CD4 count, which suggests that the observed effect was due to overcoming of other structural and individual-level barriers [Importantly, the study arm remained significant in the multivariable model controlling for clinical, demographic, and substance use variables. We may hypothesize that the intervention may have facilitated access to other health and social services, and may have promoted healthier living in ways that we did not measure, thereby leading to the reduction in non-HIV related deaths in the experimental arm.It should be noted, that participants who attained a CD4 cell count of at least 200 cells/μL continued to experience substantially elevated mortality, about 3 times higher than their HIV-negative partners in our study, and up to 18 times higher than expected in their age-matched general population. These mortality rates cannot be directly compared to other studies of patients on ART, because person time calculation in this analysis was starting from study enrollment and not ART initiation. Nevertheless, it is evident in our study, that PWID who had unsuppressed viral load at baseline and initiated ART afterwards had several times higher death rate compared to other PLWH [The benefits of the integrated intervention were significant not only for direct recipients but also among their injection partners. One plausible mechanism of this effect could be that by reducing barriers to health care for indexes, our intervention also indirectly motivated and helped their partners to seek care, reduce injection drug use, or increase safer injection practices. Specifically, as reported before [
PMC10173611
Other predictors of mortality
depression, death
We assessed the effects of recent injecting and non-injecting substance use on the risk of death. Among indexes, mortality was lower among participants self-reporting hazardous alcohol use or any type of injecting drug use in the past 3 months, and higher among those reporting no injecting or non-injecting drug use. One possible explanation may be that the intensity of drug use among HIV-positive PWID may gradually decline before death due to deteriorating health and decreasing tolerance to substances. Among the HIV-negative partner participants, neither injecting nor non-injecting substance use was associated with mortality.In contrast to other studies, suggesting higher mortality among opioid users compared to stimulant users [Importantly, we have also found more than twice the risk of death in index participants with moderate or severe depression. The causal pathway of this association is described by multiple studies showing the negative impact of depression at each stage of HIV treatment continuum: care engagement [Despite the well-documented role of stigma in diminishing access to life-saving treatments for PWID [
PMC10173611
Limitations
depression, deaths, death
The participants in our study were recruited using targeted outreach and peer referral; therefore the sample should not be considered representative of the entire PWID population in these countries. Our participants are likely to be more closely linked to HIV prevention and treatment services, also as a result of study participation [Our algorithm of assessment of the cause of death relied on investigation by the study teams and was restricted by the lack of contact information and considerations of participant confidentiality. This have led to a substantial proportion of deaths due to unknown causes, and in some cases could also introduce a bias. Verification by three physicians should have minimized the chance of misclassification.Most of the measures included in this analysis (ART, OAT receipt, drug use, depression) are self-reported, which is prone to recall and social desirability biases. In a sub-study measuring the presence of ART drugs in blood samples [
PMC10173611
Conclusion
death
The HPTN 074 randomized controlled trial, conducted in Indonesia, Vietnam, and Ukraine, documented directly measured mortality rates in PWID. The results confirm that both HIV-infected and uninfected PWID remain at a starkly elevated risk of death compared to the general population of the same age. Mortality related to HIV and other causes can be significantly reduced by scaling-up ART and OAT. Access to these life-saving treatments can be effectively improved by flexible integrated interventions, such as the one developed and tested in HPTN 074. Future research may explore strategies that facilitate implementation and scale-up of such interventions.
PMC10173611
Acknowledgements
We thank Myron S Cohen for his assistance in the earliest stages of this study, including its conceptualisation and securing HIV Prevention Trials Network (HPTN) funding. We also thank Wafaa M El-Sadr for her support throughout the study and critical review of the manuscript. We thank Katherine Davenny for her advocacy and assistance in the entire study process. We thank FHI 360 for their crucial role of in study implementation and Bonnie Dye and Jonathan Lucas in particular. Finally, we thank all of the HPTN protocol team, the site staff in Indonesia, Vietnam, and Ukraine for their dedication to the study, and the participants for their willingness to share their experiences through the study.
PMC10173611
Authors’ contributions
RS
WCM, IFH, CAL, and DSM conceived the study. WCM, IFH, CAL, DSM, KEL and VFG assisted with the study design. IFH, BH, TVH, KD, ZD, OZ, RS, SMR, CAL, DSM, KEL, VFG, EPM, PR, and WCM developed the protocol and assisted with study implementation or oversight. CAL, DSM, KEL, and VFG developed the intervention. BH and XG analyzed the data. KD drafted the manuscript and all authors edited, reviewed, and approved the final manuscript.
PMC10173611
Funding
AIDS
INFECTIOUS DISEASES, AIDS, ABUSE, ALLERGY
This work was supported by the National Institute of Allergy and Infectious Diseases (NIAID), the National Institute of Mental Health (NIMH), and the National Institute on Drug Abuse (NIDA) of the National Institutes of Health (NIH); award numbers UM1AI068619 [HPTN Leadership and Operations Center], UM1AI068617 [HPTN Statistical and Data Management Center], UM1AI068613 [HPTN Laboratory Center], and the University of North Carolina at Chapel Hill Center for AIDS Research (P30 AI50410). The content is solely the responsibility of the authors and does not necessarily represent the official views of the NIH.National Institutes of Health.
PMC10173611
Availability of data and materials
The datasets supporting the conclusions of this article and respective data dictionaries are included within the additional files.
PMC10173611
Declarations
PMC10173611
Ethics approval and consent to participate
All study procedures were carried out in accordance with relevant guidelines and regulations. Written informed consent was obtained from all participants enrolled in HPTN 074. The study protocol was approved by the following institutional review boards: Ukrainian Institute on Public Health Policy IRB#1 (Ukraine); Ethical Review Board for Biomedical Research Hanoi School of Public Health (Vietnam); Ethics Committee of Faculty of Medicine, University of Indonesia/Cipto Mangunkusumo Hospital (Indonesia); and the Institutional Review Board at the University of North Carolina-Chapel Hill.
PMC10173611
Consent for publication
Not applicable.
PMC10173611
Competing interests
This study was supported by the National Institutes of Health (NIH) and multiple investigators received grants or support from NIH (IFH, BH, TVH, CAL, DSM, KEL, VFG, EPM, PR, and WCM). All other authors declare no competing interests.
PMC10173611
References
PMC10173611
BACKGROUND:
stroke, acute stroke therapies, upper limb motor impairment
STROKE
Despite improvements in acute stroke therapies and rehabilitation strategies, many stroke patients are left with long-term upper limb motor impairment. We assessed whether an inhibitory repetitive transcranial magnetic stimulation treatment paradigm started within 3 weeks after stroke onset promotes upper limb motor recovery.
PMC10358447
METHODS:
stroke, upper limb motor impairment, intracerebral hemorrhage, ischemic stroke
STROKE, CORTEX, INTRACEREBRAL HEMORRHAGE, ISCHEMIC STROKE
We performed a single-center randomized, sham-controlled clinical trial. Patients with ischemic stroke or intracerebral hemorrhage and unilateral upper limb motor impairment were randomized to 10 daily sessions of active or sham continuous theta-burst stimulation (cTBS) of the contralesional primary motor cortex combined with standard upper limb therapy, started within 3 weeks after stroke onset. The primary outcome was the change in the Action Research Arm Test score from baseline (pretreatment) at 3 months after stroke. Secondary outcomes included the score on the modified Rankin Scale at 3 months and the length of stay at the rehabilitation center. Statistical analyses were performed using mixed models for repeated measures.
PMC10358447
RESULTS:
poststroke
We enrolled 60 patients between April 2017 and February 2021, of whom 29 were randomized to active cTBS and 31 to sham cTBS. One patient randomized to active cTBS withdrew consent before the intervention and was excluded from the analyses. The mean difference in the change in Action Research Arm Test score from baseline at 3 months poststroke was 9.6 points ([95% CI, 1.2–17.9];
PMC10358447
CONCLUSIONS:
stroke, disability
STROKE, CORTEX
Ten daily sessions of cTBS of the contralesional primary motor cortex combined with upper limb training, started within 3 weeks after stroke onset, promote recovery of the upper limb, reduce disability and dependence and leads to earlier discharge from the rehabilitation center.
PMC10358447
REGISTRATION:
stroke, Upper limb motor impairment, ischemic stroke
INTRACEREBRAL HAEMORRHAGE, STROKE, ISCHEMIC STROKE
URL: Upper limb motor impairment is one of the most frequent long-term neurological consequences of ischemic stroke or intracerebral haemorrhage.Spontaneous recovery after stroke is believed to be driven by neurobiological processes that occur mainly during a period of heightened brain plasticity in the first 3 months after stroke.An interhemispheric imbalance may be restored through excitation of the lesioned M1 or inhibition of the contralesional M1.Conventional LF rTMS treatment consists of 15-minute sessions, whereas the continuous theta-burst stimulation (cTBS), a novel inhibitory rTMS paradigm, has a much shorter treatment duration of 40 seconds per session. Therefore, the use of cTBS can improve patient comfort and increase cost-effectiveness of the intervention.
PMC10358447
METHODS
PMC10358447
Study Design
We performed a single-center, prospective, randomized, sham-controlled clinical trial with a single-blind intervention and a double-blind primary outcome evaluation at rehabilitation center De Hoogstraat (Utrecht, the Netherlands), according to the CONSORT (Consolidated Standards of Reporting Trials) guidelines. Deidentified participant data are available from the corresponding author upon reasonable request. A summary of the trial protocol has been published
PMC10358447
Participants
paresis, first-ever ischemic stroke, intracerebral hemorrhage
PARESIS, INTRACEREBRAL HEMORRHAGE
We included patients aged ≥18 years with first-ever ischemic stroke or intracerebral hemorrhage and a paresis of 1 arm, as defined by a Motricity Index
PMC10358447
Randomization and Masking
Patients were randomly assigned to 10 daily sessions of contralesional cTBS or to sham cTBS during 2 weeks, in addition to regular care upper limb therapy, using a secured online allocation system (Research Online V2.0, Julius Centre, the Netherlands) by the investigator performing the treatment. Randomization was stratified according to the ability to extend 1 or more fingers of the paretic arm.
PMC10358447
Procedures
CARAS, Stroke
STROKE, BRAIN
Treatment was delivered in 10 daily sessions on consecutive working days. cTBS was delivered over the contralesional M1, which was defined as the position on the scalp at which motor-evoked potentials (MEPs) with the largest peak-to-peak amplitude could be evoked in the contralateral first dorsal interosseous (FDI) muscle by delivering TMS pulses and monitoring the electromyogram. A Neuro-MS/D advanced therapeutic magnetic stimulator and an angulated 100 mm figure-of-eight TMS coil (Neurosoft, Ivanovo, Russia) were used for stimulation. Electromyogram was recorded, amplified, and digitized at a sampling frequency of 20 kHz using a 4-channel Neuro-MEP amplifier (Neurosoft, Ivanovo, Russia). cTBS consisted of continuous delivery of 3 stimuli bursts at 50 Hz repeated at 5 bursts per second for a duration of 40 seconds with a biphasic TMS-induced current at 45° to the midline. Stimulation intensity was set at 70% of the RMT. TMS coil placement was recorded using a neuronavigation system (Brain Science Tools BV, De Bilt, the Netherlands) starting from the 16th patient. cTBS or sham cTBS was delivered 15 minutes before standard upper limb therapy, which consisted of a 60-minute group therapy session of individualized upper limb exercises, according to the Concise Arm and Hand Rehabilitation Approach in Stroke (CARAS).
PMC10358447
Outcomes
poststroke, stroke, Stroke
STROKE, SECONDARY, STROKE
The primary outcome measure was the change in the Action Research Arm Test (ARAT) score from baseline at 3 months after stroke, as recommended by the Stroke Recovery and Rehabilitation Roundtable.Predefined secondary outcomes included the change in ARAT score <12 hours, 1 week, and 1 month posttreatment, and 6 and 12 months poststroke. Other predefined secondary outcomes were tests that assess different domains of the international classification of functioning, disability, and health framework.
PMC10358447
Statistical Analysis
SECONDARY
The sample size calculation was based on an effect size of 0.55 on the ARAT score obtained from a meta-analysis.We followed the prespecified statistical analysis plan, which is available in the We performed similar analyses for the effect of treatment on the secondary outcomes at all visits except for the mRS and length of stay. The mRS was analyzed using a cumulative link mixed model of the total mRS scores due to the ordinal nature of the data. The effect of treatment on the length of stay and the dose of upper limb therapy and self-practice that patients received were analyzed with independent samples Statistical analysis was performed with R 4.1 and SPSS 26.0 (IBM, Chicago, IL). All hypotheses were tested a 2-sided alpha of 0.05.
PMC10358447
DISCUSSION
Headache, anxiety, disability and dependency, stroke, pain, headache/nausea, poststroke
ADVERSE EVENTS, STROKE
Patients who received active cTBS showed greater improvement in upper limb recovery as measured with the ARAT at 3 months poststroke compared with patients who received sham cTBS. The mean additional improvement of 9.5 points (17% of the maximum ARAT score) on the ARAT is clinically meaningful and exceeds the previously established minimal clinically important difference of 5.7 points (10% of the maximum ARAT score).Patients who received active cTBS showed more improvement on metrics of disability and dependency (mRS) and quality of life (upper limb section of the stroke impact scale ) at 1 month posttreatment and at 3 months poststroke, compared with patients who were treated with sham stimulation. The EuroQol-5D was a quality of life metric that did not show a treatment effect, presumably due to involvement of factors that are not directly related to an improvement in upper limb recovery, such as pain and anxiety. In addition to a treatment effect on these metrics, the length of stay in the rehabilitation center, an indicator of independency, was 18 days shorter in patients treated with active cTBS compared with patients who received sham cTBS. These findings suggest that patients who received active cTBS were able to independently perform activities of daily living and participate in society at an earlier stage compared with patients who received sham cTBS.Equally important to efficacy, the investigated cTBS treatment was safe and tolerable, as no serious adverse events were reported. Headache was more prevalent in the active cTBS group, but overall mild side-effects (headache/nausea) were uncommon (<4% of cases).We did not detect an effect of active cTBS on contralesional M1 excitability, although a trend toward long-lasting (days) inhibition of the contralesional M1 could be observed. We speculate that active cTBS leads to a short-lasting reduction (<2 hours) in contralesional M1 excitability, which has dissipated the next day, but which facilitates the effects of upper limb therapy directly following cTBS. Unfortunately, the acute effect of cTBS could not be measured as the cTBS treatments were directly followed by upper limb therapy. Our findings are in line with a recent meta-analysis that showed that contralesional inhibitory rTMS improves upper limb recovery on the FMA arm score with a similar mean difference of 9 points at 3 months poststroke.Earlier meta-analyses of effects of rTMS treatment within the first months poststroke were based (almost) exclusively on inhibition with conventional LF rTMS.A third of the screened patients did not meet the inclusion criteria because the treatment could not be started within 3 weeks after stroke onset. Extension of the treatment time window would have increased the number of eligible patients, but posed the risk of reducing the treatment efficacy, as a previous meta-analysis demonstrated efficacy for treatment only when started within the first month poststroke.
PMC10358447
Limitations
SECONDARY
Our study had some limitations. First, the researchers were not blinded to the treatment allocation due to practical reasons concerning the sham condition, what could have introduced bias during the treatment period. In addition, although we performed double-blinded assessment of the primary outcome at 3 months, most secondary outcomes were assessed in a single-blind fashion. However, the considerably shorter length of stay in the rehabilitation center strongly suggests that the benefits observed on the other secondary outcomes are real.Second, the sham condition did not consist of electrical stimulation to mask sensory sensations on the scalp evoked by peripheral nerve stimulation during TMS, and successful blinding of patients was not verified by asking patients which treatment group they believed they were assigned to. However, all patients were naive to cTBS treatment, which potentially reduces the bias introduced by limitations in the sham condition.Third, we only measured the duration of upper limb therapy that patients received during the 2-week treatment period and the week thereafter. However, it is unlikely that patients in both groups received different durations of upper limb therapy after this period, as all patients were part of the same treatment schedule. While upper limb therapy content was balanced between groups at onset of treatment, this may have changed based on the additional improvement in upper limb function due to active cTBS, in accordance with the CARAS protocol. However, because both patients and therapists were blinded to treatment allocation, we consider it unlikely that patients in the active cTBS group received different upper limb therapy content unless the change in therapy was the direct result of improved motor function caused by active cTBS.Fourth, we only scored the upper limb component of the FMA. Assessment of the lower limb component could have provided insights into the specificity of the treatment.Fifth, the investigated sample consisted predominantly of males, which could limit generalizability of the results to females.Finally, our treatment paradigm was based on the theoretical interhemispheric imbalance model. However, we did not explicitly evaluate the interhemispheric balance before or after the intervention.
PMC10358447
Future Research
stroke
STROKE
The results in this single-center study are promising and provide a strong foundation for future multicenter trials to provide conclusive evidence on the efficacy of cTBS treatment in the promotion of upper limb recovery after stroke. These trials could tailor treatment to individual patients, as recent studies suggest that efficacy of contralesional cTBS may depend on stroke severity
PMC10358447
Conclusions
stroke
STROKE
In the present study, treatment with cTBS of the contralesional M1 combined with upper limb training, started within 3 weeks after stroke onset, improved upper limb motor recovery and led to better functional outcomes. Some treatment benefits persisted up to at least 12 months after stroke.
PMC10358447
ARTICLE INFORMATION
PMC10358447
Acknowledgments
Stroke
STROKE, BRAIN
The authors thank all patients and investigators for their contribution to the B-STARS study (Brain Stimulation for Arm Recovery After Stroke).
PMC10358447
Sources of Funding
BRAIN
This work was supported by the Netherlands Organization for Scientific Research (VICI 016.130.662) and in part by Brain Science Tools B.V. The funders had no role in study design, data collection, data analysis, data interpretation, or writing of the article.
PMC10358447
Disclosures
HEART, DER, BRAIN
J.J.T. Vink is a part-time employee of and Dr Neggers is the CEO and shareholder of Brain Science Tools B.V. Data analysis was performed during J.J.T. Vink’s part-time employment at Brain Science Tools B.V. Dr van der Worp is consultant for Bayer and receives funding from the Dutch Heart Foundation, Horizon 2020 Framework Programme and Stryker. The other authors report no conflicts.
PMC10358447
SUPPLEMENTAL MATERIAL
Tables S1–S2Trial protocolStatistical analysis planCONSORT checklist
PMC10358447
Supplementary Material
PMC10358447
Nonstandard Abbreviations and Acronyms
Action Research Arm TestConcise Arm and Hand Rehabilitation Approach in Strokecontinuous theta-burst stimulationFugl-Meyer Assessmentlow-frequencyprimary motor cortexmodified Rankin Scaleresting motor thresholdrepetitive transcranial magnetic stimulationJ.J.T. Vink and E.C.C. van Lieshout contributed equally.For Sources of Funding and Disclosures, see page 1940.Supplemental Material is available at
PMC10358447
REFERENCES
PMC10358447
Subject terms
diaphragmatic dyskinesias, Parkinson’s disease, PD
AIRWAY OBSTRUCTION
Upper airway obstruction, reduced maximal expiratory and inspiratory flows, reduced lung volumes, abnormal ventilatory control, and diaphragmatic dyskinesias are reported in patients with Parkinson’s disease (PD). Inspiratory muscle training (IMT) has been reported to be effective in improving respiratory functions; however, no studies have compared the effects of the incentive spirometer (IS) with the threshold inspiratory muscle trainer (TIMT) in patients with PD. The study aimed to compare the effects of IS and TIMT on maximum inspiratory pressure (MIP), 6-min walk distance (6-MWD), forced vital capacity (FVC), forced expiratory volume in 1 s (FEV1), and peak expiratory flow rate (PEFR) in patients with stage 1–3 according to the Hoehn and Yahr scale. 18 patients were randomly assigned to two groups, i.e., incentive spirometer (IS) and threshold inspiratory muscle trainer (TIMT) group. The IS group received IMT with volume-based IS, and the TIMT group received IMT with TIMT. MIP, 6-MWD, FVC, FEV1, and PEFR were measured before and after six weeks of training. In IS group: A significant increase (
PMC9925741
Introduction
respiratory problems, Parkinson's disease, tremors, respiratory dysfunction, PD, muscle rigidity
DISEASE, PARKINSON'S DISEASE, RESPIRATORY COMPLICATIONS, NEURODEGENERATIVE DISORDER, COMPLICATIONS
Parkinson's disease (PD) is a neurodegenerative disorder that is progressive and characterized by muscle rigidity, tremors, postural instability, bradykinesia, and autonomic dysfunctionsTherefore, treating respiratory problems in PD should be part of disease management to avoid complications in the respiratory system. However, no fixed respiratory protocols are suggested for respiratory problems as an intervention in patients with PD. According to a systematic review by Rodríguez et al.A few available types of equipment can be used as part of respiratory therapy that can improve ventilation and target lung expansion in patients with PD, thus preventing and treating respiratory complications. A volume-based incentive spirometer (IS) is a mechanical breathing device used in patients with respiratory dysfunction to aid in lung expansionAnother commonly used device for IMT is the threshold inspiratory muscle trainer (TIMT)Both devices (IS and TIMT) are used for IMT and use different mechanisms for their functioning. IS is a volume-based loading device, and TIMT is a pressure-based loading device; therefore, their effects may differ. Choosing the best from them will be an advantage for PD patients. No study has compared the effects of IS with the effects of TIMT when used for IMT in patients with PD. Therefore, the study aimed to compare the effectiveness of IS versus TIMT on maximum inspiratory pressure (MIP), 6-min walk distance (6-MWD), and pulmonary functions [forced vital capacity (FVC), forced expiratory volume in 1 s (FEV1), and peak expiratory flow rate (PEFR)] in patients with PD. We hypothesized that there is a significant difference between the effects of IS and the effects of TIMT on MIP, 6-MWD, FVC, FEV1, and PEFR in patients with PD.
PMC9925741
Methods
PMC9925741
Study design
Two arms pretest–posttest experimental research design was used with random allocation of participants into two groups, i.e., the Incentive spirometer (IS) group, and the threshold inspiratory muscle trainer (TIMT) group.
PMC9925741
Instrumentation
LUNG
PHILIPS Respironics Threshold IMT Lung Adjustable Constant Pressure Muscle Trainer (Threshold IMT, Philips Respironics, Inc., Murrysville, PA, USA)PORTEX Coach 2 incentive spirometer (Smiths Medical)Computerized Pulmonary Function Test Machine (RMS Helios 401, India)Portable MicroRPM (Respiratory Pressure Meter) (ICC 0.86–0.90)
PMC9925741