Exercise and Rehabilitation in People With Ehlers-Danlos Syndrome: A Systematic Review

Stephanie Buryk-Iggers, MSc; Nimish Mittal, MD, MSc; Daniel Santa Mina, RKin, PhD; Scott C. Adams, RKin, PhD; Marina Englesakis, MLIS; Maxim Rachinsky, MD; Laura Lopez-Hernandez, MSc, PT; Laura Hussey, MSc, PT; Laura McGillis, MN, RN(EC); Lianne McLean, RN, MN; Camille Laflamme, MD; Dmitry Rozenberg, MD, PhD; Hance Clarke, MD, PhD

Archives of Rehabilitation Research and Clinical Translation (Jun 2022)

Exercise and Rehabilitation in People With Ehlers-Danlos Syndrome: A Systematic Review

Stephanie Buryk-Iggers, MSc,
Nimish Mittal, MD, MSc,
Daniel Santa Mina, RKin, PhD,
Scott C. Adams, RKin, PhD,
Marina Englesakis, MLIS,
Maxim Rachinsky, MD,
Laura Lopez-Hernandez, MSc, PT,
Laura Hussey, MSc, PT,
Laura McGillis, MN, RN(EC),
Lianne McLean, RN, MN,
Camille Laflamme, MD,
Dmitry Rozenberg, MD, PhD,
Hance Clarke, MD, PhD

Affiliations

Stephanie Buryk-Iggers, MSc: Department of Kinesiology and Physical Education, University of Toronto, Toronto, Ontario, Canada; GoodHope Ehlers Danlos Syndrome Clinic, Toronto General Hospital, Toronto, Ontario, Canada
Nimish Mittal, MD, MSc: Department of Kinesiology and Physical Education, University of Toronto, Toronto, Ontario, Canada; GoodHope Ehlers Danlos Syndrome Clinic, Toronto General Hospital, Toronto, Ontario, Canada; Faculty of Medicine, Division of Physical Medicine and Rehabilitation, University of Toronto, Toronto, Ontario, Canada
Daniel Santa Mina, RKin, PhD: Department of Kinesiology and Physical Education, University of Toronto, Toronto, Ontario, Canada; GoodHope Ehlers Danlos Syndrome Clinic, Toronto General Hospital, Toronto, Ontario, Canada; Department of Anaesthesia and Pain Management, University Health Network, Toronto, Ontario, Canada; Corresponding author Daniel Santa Mina, RKin, PhD, Faculty of Kinesiology and Physical Education, University of Toronto, 55 Harbord S, Toronto, Ontario M5S 2W6.
Scott C. Adams, RKin, PhD: Department of Kinesiology and Physical Education, University of Toronto, Toronto, Ontario, Canada; Department of Cardiology, Toronto General Hospital Research Institute, Toronto, Ontario, Canada; Ted Rogers Cardiotoxicity Prevention Program, Peter Munk Cardiac Centre, Toronto, Ontario, Canada
Marina Englesakis, MLIS: Library and Information Services Toronto General Hospital, Toronto, Ontario, Canada
Maxim Rachinsky, MD: GoodHope Ehlers Danlos Syndrome Clinic, Toronto General Hospital, Toronto, Ontario, Canada; Department of Anaesthesia and Pain Management, University Health Network, Toronto, Ontario, Canada
Laura Lopez-Hernandez, MSc, PT: GoodHope Ehlers Danlos Syndrome Clinic, Toronto General Hospital, Toronto, Ontario, Canada
Laura Hussey, MSc, PT: GoodHope Ehlers Danlos Syndrome Clinic, Toronto General Hospital, Toronto, Ontario, Canada
Laura McGillis, MN, RN(EC): GoodHope Ehlers Danlos Syndrome Clinic, Toronto General Hospital, Toronto, Ontario, Canada
Lianne McLean, RN, MN: GoodHope Ehlers Danlos Syndrome Clinic, Toronto General Hospital, Toronto, Ontario, Canada
Camille Laflamme, MD: GoodHope Ehlers Danlos Syndrome Clinic, Toronto General Hospital, Toronto, Ontario, Canada; Faculty of Medicine, Division of Physical Medicine and Rehabilitation, University of Toronto, Toronto, Ontario, Canada
Dmitry Rozenberg, MD, PhD: Faculty of Medicine, Division of Physical Medicine and Rehabilitation, University of Toronto, Toronto, Ontario, Canada; Department of Medicine, Respirology, Toronto General Hospital Research Institute, Toronto, Ontario, Canada
Hance Clarke, MD, PhD: GoodHope Ehlers Danlos Syndrome Clinic, Toronto General Hospital, Toronto, Ontario, Canada; Faculty of Medicine, Division of Physical Medicine and Rehabilitation, University of Toronto, Toronto, Ontario, Canada; Department of Anaesthesia and Pain Management, University Health Network, Toronto, Ontario, Canada

Journal volume & issue: Vol. 4, no. 2
p. 100189

Abstract

Read online

Objective: To conduct a systematic review examining the effect of exercise and rehabilitation in people with Ehlers-Danlos syndrome (EDS). Data Sources: The following databases were systematically searched: MEDLINE, MEDLINE In-Process/ePubs, Embase, Cochrane Central Register of Controlled Trials, PsycINFO, and Cumulative Index to Nursing and Allied Health. The final time point captured by the search is November 27, 2020. Study Selection: Eligible study designs included case-control, case-series, prospective cohort, retrospective cohort, and intervention studies of structured exercise or rehabilitation interventions. Eligible populations included adults (18 years or older) with EDS (all subtypes) and hypermobility spectrum disorders. The search was restricted to articles published in English. Data Extraction: Data were extracted by 2 independent reviewers. Risk of bias was assessed using the Physiotherapy Evidence Database (PEDro) scale for randomized controlled trials (RCTs) and Risk Of Bias In Nonrandomized Studies of Interventions (ROBINS-I) for non-RCTs. Reporting quality of RCTs was assessed using the Consolidated Standards for Reporting of Trials statement with the harms extension. Reporting was guided by the Preferred Reporting Items for Systematic Reviews and Meta-Analyses checklist. Data Synthesis: The search yielded 10 eligible studies including 330 participants. The study designs included 5 RCTs, 1 cohort, 2 single-arm interventions, 1 retrospective, and 1 feasibility study. All studies showed some improvement in a physical and/or psychological outcome after the intervention period. One adverse event (nonserious) potentially related to the intervention was reported. Of the 5 RCTs, 2 were rated as high quality with low risk of bias using PEDro, and the majority of non-RCTs were rated as critical risk of bias by ROBINS-I. Conclusions: The results suggest that exercise and rehabilitation may be beneficial for various physical and psychological outcomes. Adequately powered and rigorous RCTs of exercise and rehabilitation interventions for people with EDS are needed.

Published in Archives of Rehabilitation Research and Clinical Translation

ISSN: 2590-1095 (Online)
Publisher: Elsevier
Country of publisher: Netherlands
LCC subjects: Medicine: Medicine (General)
Website: https://www.journals.elsevier.com/archives-of-rehabilitation-research-and-clinical-translation

About the journal

Abstract

Keywords