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Manns S.1, Cramp F.1, Lewis R.2, Clark E.3, Palmer S.1
1University of the West of England, Allied Health Professions, Bristol, United Kingdom, 2North Bristol NHS Trust, Bristol, United Kingdom, 3University of Bristol, Musculoskeletal Research Unit, Bristol, United Kingdom
Background: Joint Hypermobility Syndrome (JHS) is characterised by multiple joint laxity in association with pain, fatigue, proprioception deficits, repeated cycles of injury and a range of psychological sequelae (Terry et al 2015, Smith et al 2014). JHS is likely to be very common in musculoskeletal practice, with 30% of patients referred to a musculoskeletal triage service in the United Kingdom (UK) meeting the diagnostic criteria (Connelly et al 2015). The Bristol Impact of Hypermobility (BIoH) questionnaire is a condition-specific patient-reported outcome measure developed with adults with JHS (Palmer et al 2006). It gives a maximum score of 360, with higher scores representing more severe impact. The BIoH questionnaire has strong concurrent validity (r=-0.725, n=615) with the physical component score of the Short Form 36 questionnaire (Palmer et al 2006) however other important psychometric properties have yet to be established. Further validation is required to support its use in clinical research and practice.
Purpose: This study aimed to evaluate the appropriateness, validity, acceptability, feasibility and interpretability (Fitzpatrick et al 1998) of the BIoH questionnaire from two perspectives - people with JHS and physiotherapists with experience of treating people with JHS.
Methods: Adults with JHS were recruited through the Hypermobility Syndromes Association (HMSA), a patient organisation in the United Kingdom. Patients were purposively selected on the basis of gender, age and condition severity (based on their BIoH score). Physiotherapists with a professional interest in JHS were recruited through the HMSA and an online professional network. A qualitative evaluation using semi-structured telephone interviews was employed. Interviews incorporated a think aloud exercise and additional prompts to explore the questionnaires properties. Interviews were transcribed and data synthesised using a framework matrix.
Results: Eleven patients (n=9 women, median age 48 years, median BIoH score 238) and nine physiotherapists (n=9 women, median time since qualification 30 years) took part in interviews. 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. Items highlighted for potential future inclusion on the questionnaire included using hand-held technology and hair washing/drying. Interestingly, questions about keyboard use and hair washing were excluded during initial development of the questionnaire as they were rated as relatively unimportant. The value of including a question about intimacy was identified by a physiotherapist and positively received by patients.
Conclusion(s): Patients and physiotherapists regarded the BIoH questionnaire as a welcome addition to the toolkit available to support those with JHS. It was broadly accepted as reflecting the experience of people with JHS in sufficient detail to support management of the condition.
Implications: The findings further support the validity of the BIoH questionnaire, increasing confidence in using it to support future clinical research and practice in this under-recognised and poorly managed condition.
Funding acknowledgements: Faculty of Health & Applied Sciences, University of the West of England, Bristol, UK.
Topic: Musculoskeletal
Ethics approval: Faculty of Health & Applied Sciences Ethics Sub-Committee, University of the West of England, Bristol (HAS/15/01/99).
All authors, affiliations and abstracts have been published as submitted.
