{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,26]],"date-time":"2026-09-26T10:02:05Z","timestamp":1790416925371,"version":"4.1.0"},"reference-count":103,"publisher":"National Institute for Health and Care Research","issue":"47","license":[{"start":{"date-parts":[[2016,6,1]],"date-time":"2016-06-01T00:00:00Z","timestamp":1464739200000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/www.nationalarchives.gov.uk\/doc\/non-commercial-government-licence\/non-commercial-government-licence.htm"}],"funder":[{"DOI":"10.13039\/501100000664","name":"Health Technology Assessment programme","doi-asserted-by":"publisher","award":["10\/98\/05"],"award-info":[{"award-number":["10\/98\/05"]}],"id":[{"id":"10.13039\/501100000664","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Health Technol Assess"],"abstract":"<jats:sec id=\"abs1-1\"><jats:title>Background<\/jats:title><jats:p>Joint hypermobility syndrome (JHS) is a heritable disorder associated with laxity and pain in multiple joints. Physiotherapy is the mainstay of treatment, but there is little research investigating its clinical effectiveness.<\/jats:p><\/jats:sec><jats:sec id=\"abs1-2\"><jats:title>Objectives<\/jats:title><jats:p>To develop a comprehensive physiotherapy intervention for adults with JHS; to pilot the intervention; and to conduct a pilot randomised controlled trial (RCT) to determine the feasibility of conducting a future definitive RCT.<\/jats:p><\/jats:sec><jats:sec id=\"abs1-3\"><jats:title>Design<\/jats:title><jats:p>Patients\u2019 and health professionals\u2019 perspectives on physiotherapy for JHS were explored in focus groups (stage 1). A working group of patient research partners, clinicians and researchers used this information to develop the physiotherapy intervention. This was piloted and refined on the basis of patients\u2019 and physiotherapists\u2019 feedback (stage 2). A parallel two-arm pilot RCT compared \u2018advice\u2019 with \u2018advice and physiotherapy\u2019 (stage 3). Random allocation was via an automated randomisation service, devised specifically for the study. Owing to the nature of the interventions, it was not possible to blind clinicians or patients to treatment allocation.<\/jats:p><\/jats:sec><jats:sec id=\"abs1-4\"><jats:title>Setting<\/jats:title><jats:p>Stage 1 \u2013 focus groups were conducted in four UK locations. Stages 2 and 3 \u2013 piloting of the intervention and the pilot RCT were conducted in two UK secondary care NHS trusts.<\/jats:p><\/jats:sec><jats:sec id=\"abs1-5\"><jats:title>Participants<\/jats:title><jats:p>Stage 1 \u2013 patient focus group participants (<jats:italic>n<\/jats:italic>\u2009=\u200925, three men) were aged &gt;\u200918 years, had a JHS diagnosis and had received physiotherapy within the preceding 12 months. The health professional focus group participants (<jats:italic>n<\/jats:italic>\u2009=\u200916, three men; 14 physiotherapists, two podiatrists) had experience of managing JHS. Stage 2 \u2013 patient participants (<jats:italic>n<\/jats:italic>\u2009=\u20098) were aged &gt;\u200918 years, had a JHS diagnosis and no other musculoskeletal conditions causing pain. Stage 3 \u2013 patient participants for the pilot RCT (<jats:italic>n<\/jats:italic>\u2009=\u200929) were as for stage 2 but the lower age limit was 16 years.<\/jats:p><\/jats:sec><jats:sec id=\"abs1-6\"><jats:title>Intervention<\/jats:title><jats:p>For the pilot RCT (stage 3) the advice intervention was a one-off session, supplemented by advice booklets. All participants could ask questions specific to their circumstances and receive tailored advice. Participants were randomly allocated to \u2018advice\u2019 (no further advice or physiotherapy) or \u2018advice and physiotherapy\u2019 (an additional six 30-minute sessions over 4 months). The physiotherapy intervention was supported by a patient handbook and was delivered on a one-to-one patient\u2013therapist basis. It aimed to increase patients\u2019 physical activity through developing knowledge, understanding and skills to better manage their condition.<\/jats:p><\/jats:sec><jats:sec id=\"abs1-7\"><jats:title>Main outcome measures<\/jats:title><jats:p>Data from patient and health professional focus groups formed the main outcome from stage 1. Patient and physiotherapist interview data also formed a major component of stages 2 and 3. The primary outcome in stage 3 related to the feasibility of a future definitive RCT [number of referrals, recruitment and retention rates, and an estimate of the value of information (VOI) of a future RCT]. Secondary outcomes included clinical measures (physical function, pain, global status, self-reported joint count, quality of life, exercise self-efficacy and adverse events) and resource use (to estimate cost-effectiveness). Outcomes were recorded at baseline, 4 months and 7 months.<\/jats:p><\/jats:sec><jats:sec id=\"abs1-8\"><jats:title>Results<\/jats:title><jats:p>Stage 1 \u2013 JHS is complex and unpredictable. Physiotherapists should take a long-term holistic approach rather than treating acutely painful joints in isolation. Stage 2 \u2013 a user-informed physiotherapy intervention was developed and evaluated positively. Stage 3 \u2013 recruitment to the pilot RCT was challenging, primarily because of a perceived lack of equipoise between advice and physiotherapy. The qualitative evaluation provided very clear guidance to inform a future RCT, including enhancement of the advice intervention. Some patients reported that the advice intervention was useful and the physiotherapy intervention was again evaluated very positively. The rate of return of questionnaires was low in the advice group but reasonable in the physiotherapy group. The physiotherapy intervention showed evidence of promise in terms of primary and secondary clinical outcomes. The advice arm experienced more adverse events. The VOI analysis indicated the potential for high value from a future RCT. Such a trial should form the basis of future research efforts.<\/jats:p><\/jats:sec><jats:sec id=\"abs1-9\"><jats:title>Conclusion<\/jats:title><jats:p>A future definitive RCT of physiotherapy for JHS seems feasible, although the advice intervention should be made more robust to address perceived equipoise and subsequent attrition.<\/jats:p><\/jats:sec><jats:sec id=\"abs1-10\"><jats:title>Trial registration<\/jats:title><jats:p>Current Controlled Trials ISRCTN29874209.<\/jats:p><\/jats:sec><jats:sec id=\"abs1-11\"><jats:title>Funding<\/jats:title><jats:p>This project was funded by the National Institute for Health Research (NIHR) Health Technology Assessment programme and will be published in full in<jats:italic>Health Technology Assessment<\/jats:italic>; Vol. 20, No. 47. See the NIHR Journals Library website for further project information.<\/jats:p><\/jats:sec>","DOI":"10.3310\/hta20470","type":"journal-article","created":{"date-parts":[[2016,7,1]],"date-time":"2016-07-01T11:28:39Z","timestamp":1467372519000},"page":"1-264","update-policy":"https:\/\/doi.org\/10.3310\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":21,"title":["The feasibility of a randomised controlled trial of physiotherapy for adults with joint hypermobility syndrome"],"prefix":"10.3310","volume":"20","clinical-trial-number":[{"clinical-trial-number":"isrctn29874209","registry":"10.18810\/isrctn","type":"results"}],"author":[{"given":"Shea","family":"Palmer","sequence":"first","affiliation":[{"name":"Department of Allied Health Professions, University of the West of England, Bristol, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fiona","family":"Cramp","sequence":"additional","affiliation":[{"name":"Department of Allied Health Professions, 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UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nicky","family":"Welton","sequence":"additional","affiliation":[{"name":"Bristol Randomised Trials Collaboration, University of Bristol, Bristol, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Howard","family":"Thom","sequence":"additional","affiliation":[{"name":"Bristol Randomised Trials Collaboration, University of Bristol, Bristol, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rohini","family":"Terry","sequence":"additional","affiliation":[{"name":"Bristol Randomised Trials Collaboration, University of Bristol, Bristol, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katharine A","family":"Rimes","sequence":"additional","affiliation":[{"name":"Institute of Psychiatry, Psychology and Neuroscience, King\u2019s College London, London, 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