HOPE for Hypermobile Ehlers–Danlos Syndrome (hEDS) and Hypermobility Spectrum Disorder (HSD)—A Pilot Randomised Controlled Trial of Feasibility, Acceptability and Appropriateness

IF 3.5 2区医学 Q1 ANESTHESIOLOGY

European Journal of Pain Pub Date : 2025-04-29 DOI:10.1002/ejp.70030

Min Tze Chew, Emre Ilhan, Leslie L. Nicholson, Sarah Kobayashi, Verity Pacey, Alan Hakim, Cliffton Chan

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Abstract

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.

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hypermobilable ehers - danlos综合征（hEDS）和hypermobiledspectrum Disorder （HSD）的治疗希望——可行性、可接受性和适宜性的随机对照试验

背景：慢性广泛性疼痛的在线疼痛管理方案的可行性和有效性已经被证明；然而，目前还没有针对多动性埃勒-丹洛斯综合征（hEDS）或多动性谱系障碍（HSD）的经验评估方案。为了弥补这一差距，Hypermobile在线疼痛管理（HOPE）项目在利益相关者的参与下进行了试点测试。方法对HOPE项目进行随机对照试验，评估干预措施的可行性（FIM）、干预措施的可接受性（AIM）和干预适当性（IAM）以及8项临床疗效指标。干预参与者有8周的时间来完成12个自定进度模块，而对照组参与者继续照常治疗。通过在线调查对参与者进行基线、治疗后和治疗后3个月的评估。结果：随机招募72名受试者。两名对照组参与者在开始前退出，留下34名对照组和36名干预参与者进行分析。总体而言，81%-91%同意/完全同意HOPE方案可行，平均FIM评分为4.3/5 (SD 0.7)； 62%-76%同意/完全同意HOPE方案可接受，平均AIM评分为3.9/5 (SD 0.96)； 67%-76%同意/完全同意HOPE方案合适，平均IAM评分为4.0/5 （SD 0.9）。干预组仅有两项临床疗效指标有显著改善，干预后3个月，最严重疼痛强度（Cohen’s d = 0.63）和多动性影响（Cohen’s d = 0.32）分别有中等和较小的效果。结论HOPE方案似乎可行、可接受和适当，在最大疼痛强度和活动过度影响方面有初步改善。我们的研究结果为进一步探索这种在线疼痛干预的临床有效性提供了充分的支持。痛感是多动性埃勒-丹洛斯综合征（hEDS）或多动性谱系障碍（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。这项新型超移动在线疼痛管理（HOPE）项目的试点试验对于指导循证和利益相关者相关的在线资源的创建非常重要。有希望的结果表明，在更广泛地在hEDS/HSD人群中实施之前，进一步完善和重新测试其有效性的重要性，从而使他们能够自我管理疼痛。

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来源期刊

European Journal of Pain 医学-临床神经学

CiteScore

7.50

自引率

5.60%

发文量

163

审稿时长

4-8 weeks

期刊介绍： European Journal of Pain (EJP) publishes clinical and basic science research papers relevant to all aspects of pain and its management, including specialties such as anaesthesia, dentistry, neurology and neurosurgery, orthopaedics, palliative care, pharmacology, physiology, psychiatry, psychology and rehabilitation; socio-economic aspects of pain are also covered. Regular sections in the journal are as follows: • Editorials and Commentaries • Position Papers and Guidelines • Reviews • Original Articles • Letters • Bookshelf The journal particularly welcomes clinical trials, which are published on an occasional basis. Research articles are published under the following subject headings: • Neurobiology • Neurology • Experimental Pharmacology • Clinical Pharmacology • Psychology • Behavioural Therapy • Epidemiology • Cancer Pain • Acute Pain • Clinical Trials.