Development and initial validation of the Bristol Impact of Hypermobility (BIoH) questionnaire
Cramp, F. A.
- Publisher: Elsevier
Questionnaires | Familial | Joint | /dk/atira/pure/researchoutput/pubmedpublicationtype/D016428 | Focus groups | Joint laxity | Journal Article | Hypermobility | Interview | Validity of results
Aim of Investigation: Joint Hypermobility Syndrome (JHS) is a heritable disorder associated with excessive joint range of motion and pain in the absence of inflammatory joint disease. Symptomatic joint hypermobility has been reported to affect approximately 5% of women and 0.6% of men. Although it is a relatively common cause of musculoskeletal pain it is generally understood to\ud be under-recognised and poorly managed in clinical practice. This project had a number of related aims: 1. To identify the impact of JHS on adults with the condition; 2. To develop a questionnaire to assess its impact; 3. To establish the concurrent validity of the new questionnaire against the Short Form (36) Health Survey (SF-36). \ud Methods: Adults fulfilling the Brighton diagnostic criteria for JHS were invited to take part in focus groups and semi-structured telephone interviews to explore the impact of the condition. Focus groups and telephone interviews were recorded, transcribed verbatim and anonymised. Open coding of the transcripts was used to identify individual questionnaire items and coding was discussed in detail and verified by two researchers. Thematic analysis of the data did not progress beyond this first level of coding as the aim was limited to identification of individual items. The specific wording of individual questions and response options for the draft questionnaire were discussed in detail and agreed by a working group comprising patient research partners and members of the research team. The draft questionnaire and SF-36 were distributed by mail to 1,502 adult members of the Hypermobility Syndromes Association. Completed questionnaires informed a systematic and iterative process of question removal on the basis of low median reported severity (<40%) and high correlations with other questions (r>0.7). The total score of the finalised questionnaire was then correlated against responses for the SF-36. \ud Results: Two focus groups (n=12) and telephone interviews (n=3) were conducted during the initial qualitative phase of the project. Participants comprised 2 men and 13 women aged 18-45 years. The resultant draft questionnaire contained 94 scored items addressing issues such as pain, fatigue, physical function, anxiety, planning and management, and strength and weakness. A total of n=624 valid questionnaires were returned from members of the Hypermobility Syndromes Association (median age 39 years, 95% female). 39 questions were removed on the basis of low median reported severity and/or high correlations with other questions. The final Bristol Impact of Hypermobility (BIoH) questionnaire contains 55 scored items and gives a maximum score of 360. Correlation is strong with the SF-36 Physical Component Score (r=0.722*) but moderate with the Mental Component Score (r=0.446*) (*both p<0.05). \ud Conclusions: The BIoH questionnaire is the first condition-specific tool developed for JHS and it correlates strongly against the Physical Component Score of the SF-36. Other psychometric properties of the questionnaire such as test-retest reliability and sensitivity to change have yet to be established.