The Effectiveness of Conservative Interventions on Pain, Function, and Quality of Life in Adults with Hypermobile Ehlers-Danlos Syndrome/ Hypermobility Spectrum Disorders and Shoulder Symptoms: A Systematic Review

Objective: To synthesize the evidence on conservative interventions for shoulder symptoms in hypermobile Ehlers-Danlos Syndrome (hEDS) and hypermobility spectrum disorder (HSD). Data Sources: A literature search was conducted using data sources Medline, PEDro, CINAHL, AMED, Elsevier Scopus, and the Cochrane Library from January 1998 to June 2023. Study Selection: The review included primary empirical research on adults diagnosed with hEDS or HSD who experienced pain and/or mechanical shoulder symptoms and underwent conservative interventions. Initially, 17,565 studies were identified, which decreased to 9668 after duplicate removal. After title and abstract screening by 2 independent authors, 9630 studies were excluded. The full texts of the remaining 38 were assessed and 34 were excluded, leaving 4 articles for examination. Data Extraction: Two authors independently extracted data using a predefined extraction table. Quality assessment used the Joanna Briggs Institute checklists and the Template for Intervention Description and Replication. Data Synthesis: The review covered 4 studies with a total of 7 conservative interventions, including exercise programs, kinesiology taping, and elasticized compression orthoses. Standardized mean differences were calculated to determine intervention effects over time. The duration of interventions ranged from 48 hours to 24 weeks, showing positive effect sizes over time in the Western Ontario Shoulder Instability Index, pain levels, improved function in activities of daily living, and isometric and isokinetic strength. Small to negligible effect sizes were found for kinesiophobia during completion of exercise programs. Conclusions: Shoulder symptoms in hEDS/HSD are common, yet significant gaps in knowledge remain regarding conservative interventions, preventing optimal evidence-based application for clinicians. Further research is necessary to explore the most effective intervention types, frequencies, dosages, and delivery methods tailored to the specific requirements of this patient population. (c) 2024 The Authors. Published by Elsevier Inc. on behalf of American Congress of Rehabilitation Medicine. This is an open access article under the CC BY-NC-ND license (http:// creativecommons.org/licenses/by-nc-nd/4.0/).