The feasibility of a randomised controlled trial of physiotherapy for adults with joint hypermobility syndrome

Abstract

BackgroundJoint 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.ObjectivesTo 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.DesignPatients’ and health professionals’ 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’ and physiotherapists’ feedback (stage 2). A parallel two-arm pilot RCT compared ‘advice’ with ‘advice and physiotherapy’ (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.SettingStage 1 – focus groups were conducted in four UK locations. Stages 2 and 3 – piloting of the intervention and the pilot RCT were conducted in two UK secondary care NHS trusts.ParticipantsStage 1 – patient focus group participants (n = 25, three men) were aged > 18 years, had a JHS diagnosis and had received physiotherapy within the preceding 12 months. The health professional focus group participants (n = 16, three men; 14 physiotherapists, two podiatrists) had experience of managing JHS. Stage 2 – patient participants (n = 8) were aged > 18 years, had a JHS diagnosis and no other musculoskeletal conditions causing pain. Stage 3 – patient participants for the pilot RCT (n = 29) were as for stage 2 but the lower age limit was 16 years.InterventionFor 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 ‘advice’ (no further advice or physiotherapy) or ‘advice and physiotherapy’ (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–therapist basis. It aimed to increase patients’ physical activity through developing knowledge, understanding and skills to better manage their condition.Main outcome measuresData 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.ResultsStage 1 – JHS is complex and unpredictable. Physiotherapists should take a long-term holistic approach rather than treating acutely painful joints in isolation. Stage 2 – a user-informed physiotherapy intervention was developed and evaluated positively. Stage 3 – 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.ConclusionA 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.Trial registrationCurrent Controlled Trials ISRCTN29874209.FundingThis project was funded by the National Institute for Health Research (NIHR) Health Technology Assessment programme and will be published in full inHealth Technology Assessment; Vol. 20, No. 47. See the NIHR Journals Library website for further project information.

Understanding the psychosocial impact of joint hypermobility syndrome and Ehlers–Danlos syndrome hypermobility type: a qualitative interview study

“…Often, it was not that a treatment had worked, but that healthcare professionals had taken the time to listen compassionately and empathetically to patients "rather than Invalidation can be a common problem with rheumatic diseases [41], and involves a lack of understanding or negative social responses from others, such as disbelief, rejection, stigmatization and suspicion that the problem may be psychological in origin [41]. In line with other examples within the literature, participants in this study experienced very long waits for diagnosis, allegations of hypochondria or malingering, and a lack of understanding and knowledge of the condition from healthcare professionals [12,13,15,42,43].…”

The evidence‐based rationale for physical therapy treatment of children, adolescents, and adults diagnosed with joint hypermobility syndrome/hypermobile Ehlers Danlos syndrome

“…Education for health professionals [Billings et al, ; Rombaut et al, ; Terry et al, ; Lyell et al, ; Russek et al, ] is paramount in order to optimize physical therapy provision. A recent feasibility study of a six session package of treatment demonstrates future potential [Palmer et al, ,] and further research is required to explore the specific therapeutic actions of physical therapy for managing JHS/hEDS. Recently, a meta‐analysis revealed that for treatment of adults, a significant pain reduction was achieved by a variety of physical and cognitive approaches.…”

Ability of the Bristol Impact of Hypermobility questionnaire to discriminate between people with and without Joint Hypermobility Syndrome: a known‐group validity study

“…There has been recent interest in validating upper and lower limb assessment scales that account for a greater number of joints and planes of movement (Meyer, Chan, Hopper, & Nicholson, ; Nicolson and Chan, ) and it will be interesting to see if such scales can be incorporated into the diagnostic criteria in due course. Secondly, patients have reported that physiotherapists often only treat single joints in isolation, particularly if they have been referred with one particularly problematic joint (Palmer et al, ). It is clear that such a reductionist approach to management is unlikely to be successful and a much more holistic approach is required that considers the biomechanical relationships between body areas and addresses psychosocial factors.…”

“…This lack of knowledge and understanding of the condition has meant that assessment and treatment of JHS patients has been problematic. Both health professionals and patients have expressed a need for JHS to be treated holistically, as often only a single aspect or single joint is considered (Palmer et al, ). People with JHS present with such a wide array of signs and symptoms that they cannot be treated with one singular intervention (Malfait et al, ).…”

“…Assessment of JHS has been identified as an area where clinical practice could be improved, with a mismatch between what physiotherapists state as the aims of management and the outcome domains they assess to identify the success of management (Palmer et al, ). The Bristol Impact of Hypermobility (BIoH) questionnaire is the first condition‐specific tool to assess the impact of JHS (Palmer et al, ). It was developed in close partnership with people with JHS, researchers and clinicians.…”