Elsevier

Physiotherapy

Volume 103, Issue 2, June 2017, Pages 186-192
Physiotherapy

Development and initial validation of the Bristol Impact of Hypermobility questionnaire

https://doi.org/10.1016/j.physio.2016.04.002Get rights and content

Abstract

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.

Introduction

Joint hypermobility syndrome (JHS) is a heritable connective tissue disorder characterised by excessive joint range of motion and pain [1]. It has been reported to affect up to 5% of women and 0.6% of men [2], although there is a lack of good-quality epidemiological evidence for the true prevalence of JHS in the general population. The prevalence in musculoskeletal practice contexts is likely to be very high, however, with 30% of those referred to a musculoskeletal triage clinic in the UK meeting the Brighton diagnostic criteria [3], [4].

JHS is associated with a wide range of problems including pain; fatigue; proprioception deficits; and repeated cycles of injury, anxiety and catastrophising [5]. It may also be associated with a range of autonomic and gastrointestinal symptoms, and functional difficulties indicative of developmental coordination disorder/dyspraxia [6]. Empirical data have shown that, compared with healthy controls, JHS has a significant impact on outcomes such as exercise endurance, gait, pain, proprioception, strength, function and quality of life in both children [7], [8], [9], [10] and adults [11], [12], [13], [14]. A recent systematic review and meta-analysis confirmed the impact of JHS on a range of psychological variables such as fear, agoraphobia, anxiety, depression and panic disorders [15].

Physiotherapy, particularly exercise, is a mainstay of treatment for JHS, although recent systematic reviews highlighted the lack of research evidence [16], [17]. The trials in adults included in those reviews used a range of patient-reported outcome measures (PROMs), including the Short Form 36 (SF-36) [18], the Arthritis Impact Measurement Scales 2 (AIMS-2) [13] and a questionnaire developed by Barton and Bird [19]. Of those, only the SF-36 captured improvements following exercise [18]. Only one of the five AIMS-2 subscales changed with exercise [13], and no changes were evident in Barton and Bird's questionnaire [19]. Therefore, if exercise is effective (which has yet to be demonstrated convincingly [16]), only the SF-36 seemed to demonstrate sufficient measurement sensitivity. Closer inspection of these PROMs identified a lack of face, content and construct validity [20] for many issues reported by people with JHS [5]. For example, Barton and Bird's questionnaire [19] focused on lower limb activity (such as ascending and descending stairs, squatting, standing up and walking), failing to reflect upper limb functional difficulties. Neither the process of development nor the psychometric properties of the questionnaire were reported. A recent survey of physiotherapy practice in the UK [21] highlighted a lack of congruence between the aims of physiotherapy management for JHS and the tools used to assess the effectiveness of management. There is, therefore, a need to develop a condition-specific, psychometrically sound, outcome measure to underpin future research and clinical practice in this area.

This project had a number of related aims.

  • Stage 1 – to identify the impact of JHS on adults with the condition to inform initial patient-specific questionnaire items.

  • Stage 2 – to develop a questionnaire to assess the personal impact of JHS.

  • Stage 3 – to reduce the number of questionnaire items and establish the concurrent validity of the new questionnaire against the SF-36.

Section snippets

Method

Ethical approval was obtained from the South West 5 NHS Research Ethics Committee. The research was conducted in three stages.

  • Stage 1 – identification of questionnaire items. Methods: focus groups and telephone interviews with people with JHS.

  • Stage 2 – development of the initial questionnaire. Methods: working group of patient research partners and researchers; ‘think aloud’ evaluation.

  • Stage 3 – item reduction and validation of the questionnaire. Methods: administration of the initial

Stage 1

Stage 1 recruited 15 people with JHS and they contributed to two focus groups (both n = 6/15) and telephone interviews (n = 3/15). Of these, 13/15 (87%) were women, 2/15 (13%) were aged 18 to 25 years, 7/15 (47%) were aged 26 to 35 years, and 6/15 (40%) were aged 36 to 45 years. A wide range of issues related to the impact of JHS were raised, encompassing impairment, activity limitations and participation restrictions [25]. The issues identified included items common to many other long-term

Discussion

The new BIoH questionnaire is the first condition-specific tool validated for JHS. It was developed in close collaboration with people with JHS, and seems to be comprehensive in reflecting items of importance. Scores correlate strongly with the PCS of the SF-36, with the strongest relationship being evident with bodily pain domain scores. Correlation with the MCS of the SF-36 was much more modest. This suggests that the BIoH questionnaire predominantly captures information about physical

Conclusion

The new BIoH questionnaire has demonstrated initial potential to inform future research and clinical practice in this under-recognised and poorly managed condition.

Acknowledgements

The authors wish to acknowledge the assistance of HMSA in recruiting people with JHS, and to thank all of the participants for taking part in the research. The authors also wish to thank their patient research partners Alison Cossons and Jennifer Elvin, and Shahid Muhammad for completing questionnaire data entry. SF-36 was used under licence from OptumInsight (Licence No. QM013824).
Ethical approval: Ethical approval was obtained from the South West 5 NHS Research Ethics Committee (10/H0107/46).

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