针对可活动过度型埃勒斯-当洛综合征（hEDS）和活动过度谱系障碍（HSD）的希望——一项关于可行性、可接受性和适宜性的试点随机对照试验
HOPE for Hypermobile Ehlers-Danlos Syndrome (hEDS) and Hypermobility Spectrum Disorder (HSD)-A Pilot Randomised Controlled Trial of Feasibility, Acceptability and Appropriateness.
作者信息
Chew Min Tze, Ilhan Emre, Nicholson Leslie L, Kobayashi Sarah, Pacey Verity, Hakim Alan, Chan Cliffton
机构信息
Department of Health Sciences, Faculty of Medicine, Health and Human Sciences, Macquarie University, North Ryde, New South Wales, Australia.
Faculty of Medicine and Health, Kolling Institute, University of Sydney, St Leonards, New South Wales, Australia.
出版信息
Eur J Pain. 2025 Jul;29(6):e70030. doi: 10.1002/ejp.70030.
BACKGROUND
Feasibility and effectiveness of online pain management programs for chronic widespread pain conditions have been shown; yet, there are no empirically evaluated programs for hypermobile Ehlers-Danlos syndrome (hEDS) or hypermobility spectrum disorder (HSD). Bridging this gap, the Hypermobile Online Pain managemEnt (HOPE) program was developed with stakeholder input to be pilot tested.
METHODS
We conducted a randomised controlled trial of the HOPE program to assess Feasibility of Intervention Measure (FIM), Acceptability of Intervention Measure (AIM) and Intervention Appropriateness Measure (IAM) and eight clinical effectiveness outcomes. Intervention participants were given 8 weeks to complete 12 self-paced modules, while control participants continued treatment as usual. Participants were assessed at baseline, post-treatment and 3 months post-treatment using online surveys.
RESULTS
Seventy-two participants were recruited and randomised. Two control participants withdrew before starting, leaving 34 controls and 36 intervention participants for analysis. In all, 81%-91% agreed/completely agreed that the HOPE program was feasible with mean FIM score of 4.3/5 (SD 0.7), 62%-76% agreed/completely agreed that it was acceptable with mean AIM score of 3.9/5 (SD 0.96) and 67%-76% agreed/completely agreed that it was appropriate with mean IAM score of 4.0/5 (SD 0.9). Only two clinical effectiveness outcomes were significantly improved in the intervention group, with moderate and small effect sizes in worst pain intensity (Cohen's d = 0.63) and impact of hypermobility (Cohen's d = 0.32) at 3 months post-intervention, respectively.
CONCLUSION
The HOPE program seemed feasible, acceptable and appropriate, with preliminary improvements in worst pain intensity and impact of hypermobility. Our findings warrant a fully powered trial to further explore the clinical effectiveness of this online pain intervention.
SIGNIFICANCE STATEMENT
Pain is of high concern among people with hypermobile Ehlers-Danlos syndrome (hEDS) or hypermobility spectrum disorder (HSD), yet there are limited online pain management resources for them. This pilot trial of the novel Hypermobile Online Pain managemEnt (HOPE) program is important in guiding the creation of evidence-based and stakeholder-relevant online resources. Promising results suggest the importance of further refinement and retesting its effectiveness before wider implementation among the hEDS/HSD population so as to empower them in pain self-management.
背景
在线疼痛管理项目对慢性广泛性疼痛病症的可行性和有效性已得到证实；然而，目前尚无针对可弯曲性埃勒斯-当洛综合征（hEDS）或可弯曲性谱系障碍（HSD）的经实证评估的项目。为填补这一空白，在利益相关者的参与下开发了可弯曲性在线疼痛管理（HOPE）项目并进行试点测试。
方法
我们对HOPE项目进行了一项随机对照试验，以评估干预措施可行性（FIM）、干预措施可接受性（AIM）和干预措施适宜性（IAM）以及八项临床有效性指标。干预组参与者有8周时间完成12个自主进度的模块，而对照组参与者继续照常治疗。使用在线调查问卷在基线、治疗后和治疗后3个月对参与者进行评估。
结果
招募了72名参与者并将其随机分组。两名对照参与者在开始前退出，剩余34名对照组和36名干预组参与者进行分析。总体而言，81%-91%的人同意/完全同意HOPE项目是可行的，FIM平均得分为4.3/5（标准差0.7）；62%-76%的人同意/完全同意该项目是可接受的，AIM平均得分为3.9/5（标准差0.96）；67%-76%的人同意/完全同意该项目是适宜的，IAM平均得分为4.0/5（标准差0.9）。干预组仅有两项临床有效性指标有显著改善，干预后3个月时，在最严重疼痛强度（科恩d值=0.63）和可弯曲性影响（科恩d值=0.32）方面分别有中等和较小的效应量。
结论
HOPE项目似乎可行、可接受且适宜，在最严重疼痛强度和可弯曲性影响方面有初步改善。我们的研究结果需要进行一项充分有力的试验，以进一步探索这种在线疼痛干预的临床有效性。
意义声明
疼痛是可弯曲性埃勒斯-当洛综合征（hEDS）或可弯曲性谱系障碍（HSD）患者高度关注的问题，但针对他们的在线疼痛管理资源有限。这项新型可弯曲性在线疼痛管理（HOPE）项目的试点试验对于指导创建基于证据且与利益相关者相关的在线资源具有重要意义。有前景的结果表明，在hEDS/HSD人群中更广泛实施之前，进一步完善并重新测试其有效性以增强他们的疼痛自我管理能力非常重要。
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