title: Disability in Adolescents and Adults Diagnosed With Hypermobility-Related Disorders: A Meta-Analysis.
abstract: To (1) establish the association of the most common reported symptoms on disability; and (2) study the effectiveness of treatment on disability in patients with Ehlers-Danlos syndrome-hypermobility type (EDS-HT)/hypermobility syndrome (HMS).
  
  
  
  An electronic search (Medical Subject Headings and free-text terms) was conducted in bibliographic databases CENTRAL/MEDLINE.
  
  
  
  Comparative, cross-sectional, longitudinal cohort studies and (randomized) controlled trials including patients with HMS/EDS-HT aged ≥17 years were considered for inclusion. A class of symptoms was included when 5 publications were available. In regards to treatment (physical, cognitive interventions), only (randomized) controlled trials were considered. Surgical and medicinal interventions were excluded.
  
  
  
  Bias was assessed according to the methodological scoring tools of the Cochrane collaboration. Z-score transformations were applied to classify the extent of disability in comparison with healthy controls and to ensure comparability between studies.
  
  
  
  Initially, the electronic search yielded 714 publications, and 21 articles remained for analysis after selection. The following symptoms were included for meta-analysis: pain (n=12), fatigue (n=6), and psychological distress (n=7). Pain (r=.64, P=.021), fatigue (r=.91, P=.011), and psychological distress (r=.86, P=.018) had a significant impact on disability. Regarding treatment, a significant pain reduction was achieved by a variety of physical and cognitive approaches. Treatment effectiveness on disability was not established.
  
  
  
  Disability can affect patients with HMS/EDS-HT significantly and is highly correlated with both physical and psychological factors. Although evidence is available that physical and psychological treatment modalities can induce significant pain reduction, the evidence regarding disability reduction is lacking.
authors.affiliation: Amsterdam Center for Innovative Health Practice, Center for Applied Research, Faculty of Health, University of Applied Sciences Amsterdam, Amsterdam, The Netherlands; Department of Rehabilitation, Academic Medical Center, Amsterdam, The Netherlands. Electronic address: m.c.scheper@hva.nl.
authors.forename: Mark C
authors.initials: MC
authors.lastname: Scheper
authors.affiliation: Research Unit for Musculoskeletal Function and Physiotherapy, Institute of Sports Science and Clinical Biomechanics, University of Southern Denmark, Odense M, Denmark; Institute of Occupational Therapy, Physiotherapy and Radiography, Bergen University College, Bergen, Norway.
authors.forename: Birgit
authors.initials: B
authors.lastname: Juul-Kristensen
authors.affiliation: Department of Rehabilitation Sciences and Physiotherapy, Ghent University, Ghent, Belgium.
authors.forename: Lies
authors.initials: L
authors.lastname: Rombaut
authors.affiliation: Department of Rehabilitation Medicine, Maastricht University Medical Center, CAPHRI School for Public Health and Primary Care, Maastricht, The Netherlands; Adelante Center of Expertise in Rehabilitation and Audiology, Hoensbroek, The Netherlands; Master of Pediatric Physical Therapy, University of Applied Sciences, AVANS+, Breda, The Netherlands.
authors.forename: Eugene A
authors.initials: EA
authors.lastname: Rameckers
authors.affiliation: Department of Rehabilitation Medicine, Maastricht University Medical Center, CAPHRI School for Public Health and Primary Care, Maastricht, The Netherlands; Adelante Center of Expertise in Rehabilitation and Audiology, Hoensbroek, The Netherlands.
authors.forename: Jeanine
authors.initials: J
authors.lastname: Verbunt
authors.affiliation: Amsterdam Center for Innovative Health Practice, Center for Applied Research, Faculty of Health, University of Applied Sciences Amsterdam, Amsterdam, The Netherlands; Department of Rehabilitation, Academic Medical Center, Amsterdam, The Netherlands.
authors.forename: Raoul H
authors.initials: RH
authors.lastname: Engelbert
mesh_terms: D000293:Adolescent; D000328:Adult; D006233:Persons with Disabilities / Q000523:psychology / Q000534:rehabilitation*; D004535:Ehlers-Danlos Syndrome / Q000453:epidemiology / Q000523:psychology / Q000534:rehabilitation*; D005221:Fatigue / Q000453:epidemiology / Q000523:psychology; D006801:Humans; D007593:Joint Instability / Q000453:epidemiology / Q000523:psychology / Q000534:rehabilitation*; D051346:Mobility Limitation; D015588:Observer Variation; D010146:Pain / Q000453:epidemiology / Q000523:psychology; D013315:Stress, Psychological / Q000453:epidemiology / Q000523:psychology; D013577:Syndrome
keywords: Anxiety; Depression; Ehlers-Danlos syndrome; Fatigue; International Classification of Functioning, Disability and Health; Musculoskeletal pain; Rehabilitation