Buryk-Iggers Stephanie, Mittal Nimish, Santa Mina Daniel, Adams Scott C, Englesakis Marina, Rachinsky Maxim, Lopez-Hernandez Laura, Hussey Laura, McGillis Laura, McLean Lianne, Laflamme Camille, Rozenberg Dmitry, Clarke Hance
Department of Kinesiology and Physical Education, University of Toronto, Toronto, Ontario, Canada.
GoodHope Ehlers Danlos Syndrome Clinic, Toronto General Hospital, Toronto, Ontario, Canada.
Arch Rehabil Res Clin Transl. 2022 Mar 4;4(2):100189. doi: 10.1016/j.arrct.2022.100189. eCollection 2022 Jun.
To conduct a systematic review examining the effect of exercise and rehabilitation in people with Ehlers-Danlos syndrome (EDS).
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.
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 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.
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.
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.
进行一项系统评价，考察运动和康复治疗对埃勒斯-当洛综合征（EDS）患者的影响。
对以下数据库进行了系统检索：MEDLINE、MEDLINE在研/电子出版物、Embase、Cochrane对照试验中心注册库、PsycINFO以及护理与联合健康累积索引。检索的最终时间点为2020年11月27日。
符合条件的研究设计包括病例对照研究、病例系列研究、前瞻性队列研究、回顾性队列研究以及结构化运动或康复干预的干预性研究。符合条件的人群包括患有EDS（所有亚型）和关节过度活动谱障碍的成年人（18岁及以上）。检索限于以英文发表的文章。
由2名独立评审员提取数据。使用物理治疗证据数据库（PEDro）量表对随机对照试验（RCT）进行偏倚风险评估，对非RCT使用干预性非随机研究中的偏倚风险（ROBINS-I）进行评估。使用带有危害扩展的试验报告统一标准对RCT的报告质量进行评估。报告以系统评价和Meta分析的首选报告项目清单为指导。
检索产生了10项符合条件的研究，包括330名参与者。研究设计包括5项RCT、1项队列研究、2项单臂干预研究、1项回顾性研究和1项可行性研究。所有研究均显示干预期后在身体和/或心理结局方面有一定改善。报告了1例可能与干预相关的不良事件（不严重）。在5项RCT中，2项被评为高质量，使用PEDro评估偏倚风险较低，大多数非RCT被ROBINS-I评为具有关键偏倚风险。
结果表明，运动和康复治疗可能对各种身体和心理结局有益。需要针对EDS患者进行有足够样本量且严格的运动和康复干预RCT。
