Dataset Information

A randomised controlled trial of heavy shoulder strengthening exercise in patients with hypermobility spectrum disorder or hypermobile Ehlers-Danlos syndrome and long-lasting shoulder complaints: study protocol for the Shoulder-MOBILEX study.

ABSTRACT:

Background

Four out of five patients with hypermobility spectrum disorder (HSD) or hypermobile Ehlers-Danlos syndrome (hEDS) experience shoulder complaints including persistent pain and instability. Evidence suggests that patients with HSD/hEDS who experience knee and back complaints improve with exercise-based therapy. However, no study has focused on exercise-based treatment for the shoulder in this patient group. The potential benefits of strengthening the shoulder muscles, such as increased muscle-tendon stiffness, may be effective for patients with HSD/hEDS who often display decreased strength and increased shoulder laxity/instability. The primary aim is to investigate the short-term effectiveness of a 16-week progressive heavy shoulder strengthening programme and general advice (HEAVY) compared with low-load training and general advice (LIGHT), on self-reported shoulder symptoms, function, and quality of life.

Methods

A superiority, parallel group, randomised controlled trial will be conducted with 100 patients from primary care with HSD/hEDS and shoulder complaints (persistent pain and/or instability) for more than 3 months. Participants will be randomised to receive HEAVY (full range of motion, high load) or LIGHT (neutral to midrange of motion, low load) strengthening programme three times weekly with exercises targeting scapular and rotator cuff muscles. HEAVY will be supervised twice weekly, and LIGHT three times during the 16 weeks. The primary outcome will be between-group difference in change from baseline to 16-week follow-up in the Western Ontario Shoulder Instability Index (WOSI, 0-2100 better to worse). Secondary outcomes will include a range of self-reported outcomes covering symptoms, function, and quality of life, besides clinical tests for shoulder strength, laxity/instability, and proprioception. Outcome assessors will be blinded to group allocation. Participants will be kept blind to treatment allocation through minimal information about the intervention content and hypotheses. Primary analyses will be performed by a blinded epidemiologist.

Discussion

If effective, the current heavy shoulder strengthening programme will challenge the general understanding of prescribing low-load exercise interventions for patients with HSD/hEDS and provide a new treatment strategy. The study will address an important and severe condition using transparent, detailed, and high-quality methods to potentially support a future implementation.

Trial registration

ClinicalTrials.gov NCT03869307 . Registered on 11 March 2019.

SUBMITTER: Liaghat B

PROVIDER: S-EPMC7705859 | biostudies-literature |

REPOSITORIES: biostudies-literature

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Project description:Background:People with hypermobility spectrum disorder (HSD) are in great risk of experiencing shoulder symptoms, but evidence for treatment is sparse. Therefore, the objective was to evaluate the feasibility of 16-week shoulder strengthening programme for improving shoulder strength and function in people with HSD and shoulder symptoms for more than 3?months to inform a future randomised controlled trial (RCT). Methods:Twelve participants (11 females, 39.3 ± 13.9?years) with HSD and shoulder instability and/or pain for more than 3?months underwent a 16-week heavy shoulder strengthening exercise programme three times weekly using exercises targeting scapular and rotator cuff muscles. Primary outcomes were pre-defined research progression criteria including recruitment rate (acceptable, 6 participants/month), assessment duration (acceptable: < 120?min), participant retention (acceptable: > 80% complete intervention), training adherence (acceptable: > 75% adhere to > 36 training sessions) and adverse events (acceptable: minor events with no participants discontinuing the study), besides participant and physiotherapist feedback. Secondary treatment outcomes were assessed using the Western Ontario Shoulder Instability Index (WOSI, 0-2100 better to worse), self-reported pain, kinesiophobia and fatigue, isometric shoulder strength, besides clinical tests for instability, hypermobility, laxity, and proprioception. Results:Recruitment rate was 5.6/month, assessment duration (mean ± SD) 105 ± 9?min, retention 100%, adherence 83%, and four participants experienced short-lasting soreness or pain. Participant feedback was positive, and physiotherapists found the intervention relevant and applicable to the population. The WOSI total score showed an improvement by 51% (mean ± SD, points: baseline 1037 ± 215; Follow up 509 ± 365; mean change (95% CI), - 528 (- 738, - 318)), and participants reported reduced pain, kinesiophobia and fatigue. Shoulder strength measurements improved by 28-31% (mean change (95% CI), Nm/kg: scaption 0.51 (0.23, 0.78); internal rotation 1.32 (0.70, 1.95) and external rotation 0.89 (0.37, 1.40)), and clinical tests indicated decreased shoulder laxity/instability. Conclusions:The shoulder strengthening exercise programme was feasible and safe for people with HSD and long-lasting shoulder symptoms. A future RCT, with an improved recruitment strategy, will demonstrate whether the exercise programme is also effective in improving symptoms and muscle-tendon function in this population. Trial registration:ClinicalTrials.gov: NCT03547570. Registered on May 3, 2018.

Project description:BackgroundDyspnea is a prevalent symptom in individuals with hypermobile Ehlers-Danlos Syndrome (hEDS) and generalized hypermobility spectrum disorder (G-HSD), yet its contributors have not been identified. One known contributor to dyspnea is respiratory muscle weakness. The feasibility and effectiveness of inspiratory muscle training (IMT) in combination with standard-of-care rehabilitation (aerobic, resistance, neuromuscular stabilization, and balance and proprioception exercises) in improving respiratory muscle strength and patient-reported outcomes in patients with hEDS or G-HSD have not been evaluated.ObjectiveThis study aims to evaluate dyspnea, respiratory muscle strength, and patient-reported outcome measures (PROMs) in hEDS or G-HSD compared with healthy controls and to assess the feasibility of a randomized controlled trial of IMT and standard-of-care rehabilitation for improving respiratory muscle strength, exercise capacity, and PROMs compared with standard-of-care rehabilitation in hEDS and G-HSD.MethodsThe study will include 34 participants with hEDS or G-HSD and 17 healthy, age- and sex-matched controls to compare respiratory muscle structure and function and PROMs. After baseline assessments, participants with hEDS or G-HSD will be randomized into the intervention group and provided IMT combined with Ehlers-Danlos Syndrome standard-of-care rehabilitation or into the usual care group, and provided only standard-of-care rehabilitation for 8 weeks. The intervention group will be prescribed IMT in their home environment using the POWERbreathe K5 IMT device (POWERbreathe International Ltd). IMT will comprise 2 daily sessions of 30 breaths for 5 days per week, with IMT progressing from 20% to 60% of the baseline maximal inspiratory pressure (MIP) over an 8-week period. Feasibility will be assessed through rates of recruitment, attrition, adherence, adverse events, and participant satisfaction. The primary pilot outcome is MIP change over an 8-week period in hEDS or G-HSD. Secondary outcomes will include the evaluation of dyspnea using Medical Research Council Scale and 18-point qualitative dyspnea descriptors; diaphragmatic thickening fraction using ultrasound; respiratory muscle endurance; pulmonary function; prefrontal cortical activity using functional near-infrared spectroscopy; aerobic capacity during cardiopulmonary exercise testing; quality of life using Short Form-36; and scores from the Depression, Anxiety, and Stress scale-21. These measures will also be performed once in healthy controls to compare normative values. Multivariable regression will be used to assess the contributors to dyspnea. Paired 2-tailed t tests will be used to assess the changes in MIP and secondary measures after 8 weeks of IMT.ResultsStudy recruitment began in August 2021 and, with several disruptions owing to COVID-19, is expected to be completed by December 2023.ConclusionsThis study will provide a better understanding of the factors associated with dyspnea and the feasibility and effectiveness of IMT combined with standard-of-care rehabilitation. IMT may be a novel therapeutic strategy for improving respiratory muscle function and patient-reported outcomes in individuals with hEDS or G-HSD.Trial registrationClinicalTrials.gov NCT04972565; https://clinicaltrials.gov/ct2/show/NCT04972565.International registered report identifier (irrid)DERR1-10.2196/44832.