Shea Palmer1, Indi Davey2, Laura Oliver2, Amara Preece2, Laura Sowerby2, Sophie House2. 1. Department of Allied Health Professions, Faculty of Health & Applied Sciences, University of the West of England, Blackberry Hill, Bristol, BS16 1DD, UK. shea.palmer@uwe.ac.uk. 2. Department of Allied Health Professions, Faculty of Health & Applied Sciences, University of the West of England, Blackberry Hill, Bristol, BS16 1DD, UK.
Abstract
INTRODUCTION: 'Syndromic hypermobility' encompasses heritable connective tissue disorders such as hypermobile Ehlers-Danlos syndrome and hypermobility spectrum disorders which are characterised by excessive joint range of motion and pain. Conservative interventions such as exercise are the cornerstone of management, yet their effectiveness is unclear. AIM: To systematically appraise the effectiveness of conservative management for people with syndromic hypermobility. METHOD: A systematic online database search was conducted (AMED, BND, CINAHL Plus, MEDLINE, PEDro, PsychINFO and SportDiscus). Potential articles were assessed for eligibility by two researchers against the following criteria: adults and children with a hEDS/HSD diagnosis (or equivalent diagnosis using specific criteria); non-pharmacological or non-surgical interventions; outcomes related to pain, physical function, psychological well-being or quality of life. Controlled trials and cohort studies were included. Critical Appraisal Skills Programme checklists were used to assess methodological quality. RESULTS: Eleven studies were included, comprising eight controlled trials and three cohort studies. All studies investigated interventions that had exercise as the primary component. Three small controlled studies demonstrated superior effects of conservative management relative to a control group. However, those studies only focused on a single area of the body, only recruited women, and had no long-term follow-up. All studies reported improvements in a wide range of outcomes over time. CONCLUSION: Controlled trial evidence for the superiority of conservative management over comparators is weak. There is some evidence that people improve over time. Robust randomised controlled trial research of the long-term effectiveness of 'whole-body' (rather than individual joints or body areas) conservative management is required. Key Points • Conservative management is the cornerstone of management of syndromic hypermobility. • The review found that evidence for the effectiveness of conservative management relative to no treatment or other conservative comparators was weak. • However, there was consistent evidence for effectiveness from pre- to post-treatment. • Further robust randomised controlled trial evidence is required.
INTRODUCTION: 'Syndromic hypermobility' encompasses heritable connective tissue disorders such as hypermobile Ehlers-Danlos syndrome and hypermobility spectrum disorders which are characterised by excessive joint range of motion and pain. Conservative interventions such as exercise are the cornerstone of management, yet their effectiveness is unclear. AIM: To systematically appraise the effectiveness of conservative management for people with syndromic hypermobility. METHOD: A systematic online database search was conducted (AMED, BND, CINAHL Plus, MEDLINE, PEDro, PsychINFO and SportDiscus). Potential articles were assessed for eligibility by two researchers against the following criteria: adults and children with a hEDS/HSD diagnosis (or equivalent diagnosis using specific criteria); non-pharmacological or non-surgical interventions; outcomes related to pain, physical function, psychological well-being or quality of life. Controlled trials and cohort studies were included. Critical Appraisal Skills Programme checklists were used to assess methodological quality. RESULTS: Eleven studies were included, comprising eight controlled trials and three cohort studies. All studies investigated interventions that had exercise as the primary component. Three small controlled studies demonstrated superior effects of conservative management relative to a control group. However, those studies only focused on a single area of the body, only recruited women, and had no long-term follow-up. All studies reported improvements in a wide range of outcomes over time. CONCLUSION: Controlled trial evidence for the superiority of conservative management over comparators is weak. There is some evidence that people improve over time. Robust randomised controlled trial research of the long-term effectiveness of 'whole-body' (rather than individual joints or body areas) conservative management is required. Key Points • Conservative management is the cornerstone of management of syndromic hypermobility. • The review found that evidence for the effectiveness of conservative management relative to no treatment or other conservative comparators was weak. • However, there was consistent evidence for effectiveness from pre- to post-treatment. • Further robust randomised controlled trial evidence is required.
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