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Background: The Bristol Impact of Hypermobility (BIoH) questionnaire is a condition-specific patient-reported outcome measure developed for adults with Joint Hypermobility Syndrome (JHS). It has previously demonstrated strong concurrent validity with the Short-Form 36 health questionnaire and excellent test-retest reliability., Objectives: This study aimed to evaluate its appropriateness, validity, acceptability, feasibility and interpretability., Design: A qualitative evaluation using semi-structured telephone interviews, incorporating a 'think aloud' exercise and additional prompts., Method: Adults with JHS (n = 11) were recruited through a patient organisation and physiotherapists with a professional interest in JHS (n = 9) were recruited through the same organisation and an online professional network. Interviews were transcribed and data synthesised using a framework matrix., Findings: Patients and physiotherapists commented positively on the appropriateness, validity, acceptability and feasibility of the BIoH questionnaire. Physiotherapists assessed the interpretability of the questionnaire and commented that, whilst further information might be captured, the value of that information might be limited. The questionnaire was considered comprehensive with only a very limited number of potential missing areas related to use of hand-held technology, hair washing/drying and intimacy. Interestingly, keyboard use and hair washing were excluded during initial questionnaire development as they were rated as relatively unimportant. Intimacy was not considered a comfortable addition for all participants., Conclusions: Patients and physiotherapists regarded the BIoH questionnaire as a welcome addition to the toolkit available to assess those with JHS. It was broadly accepted as reflecting the experience of people with JHS in sufficient detail to support management., (Copyright © 2018 Elsevier Ltd. All rights reserved.)

Objective: The Bristol Impact of Hypermobility (BIoH) questionnaire is a patient-reported outcome measure developed in conjunction with adults with Joint Hypermobility Syndrome (JHS). It has demonstrated strong concurrent validity with the Short Form-36 (SF-36) physical component score but other psychometric properties have yet to be established. This study aimed to determine its test-retest reliability and smallest detectable change (SDC)., Design: A test-retest reliability study., Setting: Participants were recruited from the Hypermobility Syndromes Association, a patient organisation in the United Kingdom., Patients: Recruitment packs were sent to 1080 adults who had given permission to be contacted about research., Main Outcome Measures: BIoH and SF-36 questionnaires were administered at baseline and repeated two weeks later. An 11-point global rating of change scale (-5 to +5) was also administered at two weeks. Test-retest analysis and calculation of the SDC was conducted on 'stable' patients (defined as global rating of change -1 to +1)., Results: 462 responses were received. 233 patients reported a 'stable' condition and were included in analysis (95% women; mean (SD) age 44.5 (13.9) years; BIoH score 223.6 (54.0)). The BIoH questionnaire demonstrated excellent test-retest reliability (ICC 0.923, 95% CI 0.900-0.940). The SDC was 42 points (equivalent to 19% of the mean baseline score). The SF-36 physical and mental component scores demonstrated poorer test-retest reliability and larger SDCs (as a proportion of the mean baseline scores)., Conclusion: The results provide further evidence of the potential of the BIoH questionnaire to underpin research and clinical practice for people with JHS., (Copyright © 2017 Elsevier Ltd. All rights reserved.)

Objectives: Stage 1 - to identify the impact of joint hypermobility syndrome (JHS) on adults; Stage 2 - to develop a questionnaire to assess the impact of JHS; and Stage 3 - to undertake item reduction and establish the questionnaire's concurrent validity., Design: A mixed methods study employing qualitative focus groups and interviews (Stage 1); a working group of patients, clinicians and researchers, and 'think aloud' interviews (Stage 2); and quantitative analysis of questionnaire responses (Stage 3)., Setting: Stages 1 and 2 took place in one secondary care hospital in the UK. Members of a UK-wide patient organisation were recruited in Stage 3., Participants: In total, 15, four and 615 participants took part in Stages 1, 2 and 3, respectively. Inclusion criteria were: age ≥18 years; diagnosis of JHS; no other conditions affecting physical function; able to give informed consent; and able to understand and communicate in English., Interventions: None., Main Outcome Measures: The development of a questionnaire to assess the impact of JHS., Results: Stage 1 identified a wide range of impairments, activity limitations and participation restrictions In Stage 2, a draft questionnaire was developed and refined following 'think aloud' analysis, leaving 94 scored items. In Stage 3, items were removed on the basis of low severity and/or high correlation with other items. The final Bristol Impact of Hypermobility (BIoH) questionnaire had 55 scored items, and correlated well with the physical component score of the Short Form 36 health questionnaire (r=-0.725)., Conclusions: The BIoH questionnaire demonstrated good concurrent validity. Further psychometric properties need to be established., (Copyright © 2016 Chartered Society of Physiotherapy. Published by Elsevier Ltd. All rights reserved.)

Background: 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., Objectives: 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., Design: Patients' 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., Setting: Stage 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., Participants: Stage 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., Intervention: 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 '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 Measures: 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., Results: Stage 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., Conclusion: 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., Trial Registration: Current Controlled Trials ISRCTN29874209., Funding: This project was funded by the National Institute for Health Research (NIHR) Health Technology Assessment programme and will be published in full in Health Technology Assessment; Vol. 20, No. 47. See the NIHR Journals Library website for further project information.

Joint hypermobility syndrome (JHS) is a heritable disorder associated with excessive joint range of motion and pain in the absence of inflammatory joint disease. It is a relatively common cause of musculoskeletal pain but is generally understood to be under-recognized and poorly managed in clinical practice. The present study therefore aimed to identify how JHS is diagnosed, managed and assessed in routine physiotherapy practice. A survey tool was developed from similar physiotherapy surveys of musculoskeletal practice, a review of the literature, and consultation with researchers and clinicians. Paper copies of the final survey were sent to 201 randomly selected secondary care organizations across the UK and an electronic version was advertised through physiotherapy professional networks. A total of 66 responses (80% women) were received from physiotherapists, with a wide range of clinical experience. Only 32% of respondents reported that they had received formal training in JHS management. The Brighton diagnostic criteria for JHS were not often used (only 31% of respondents). The stated aims of physiotherapy and the specific interventions employed seem well matched, with a focus on advice, education, exercise and self-management. Although pain relief was not reported as a high priority in terms of treatment aims, pain was most often assessed as an outcome, suggesting a mismatch between what clinicians aim to achieve and what they measure. The results suggest that reported management strategies are broadly appropriate to long-term musculoskeletal conditions but additional training specific to JHS may be required, particularly in diagnosis and assessment., (Copyright © 2015 John Wiley & Sons, Ltd.)