Multidisciplinary intervention for treatment of benign joint hypermobility syndrome in childhood
| ISRCTN | ISRCTN86573140 |
|---|---|
| DOI | https://doi.org/10.1186/ISRCTN86573140 |
| Secondary identifying numbers | CPMS ID: 9366 |
- Submission date
- 18/01/2017
- Registration date
- 09/03/2017
- Last edited
- 09/01/2019
- Recruitment status
- No longer recruiting
- Overall study status
- Completed
- Condition category
- Musculoskeletal Diseases
Plain English Summary
Background and study aims
Children with hypermobile joints (joints with a greater than normal range of motion) are more likely to have joint and muscle symptoms including growing pains and pain when exercising, which can become disabling in the long term and have an impact on the child’s family, social life and schooling. Hypermobility occurs in up to 30% of children aged between 5 and 17 and accounts for a large number of referrals to paediatric rheumatology clinics, where it is called Benign Joint Hypermobility Syndrome (BJHS). It is often poorly recognised and takes a long time to diagnose. Effective treatment involves lifestyle changes, altering the patient's exercise habits, joint protection, and improving body mechanics. More evidence is required of how well these treatment strategies work and their costs (clinical and cost effectiveness). Although multidisciplinary treatment involving physiotherapists, occupational therapists, and the provision of education to parents, schools and the community are all thought to be important, the extent to which these approaches are used in clinical practice varies widely. The aim of this study is to assess the clinical and cost effectiveness of a structured multidisciplinary team approach for the treatment of childhood BJHS.
Who can participate?
Children aged between 5−16 years old with pain due to BJHS
What does the study involve?
All participants and their parents/guardians are invited to attend an appointment with a physiotherapist at the hospital, who does a physical assessment of the child and completes some questionnaires with both the child and parent. This appointment takes around 1 hour. Participants are randomly allocated into one of two groups: standard treatment or structured treatment. If the participant is in the standard treatment group the child does not receive any further treatment. He/she has already had an appointment with the paediatrician and has been given information and advice about BJHS and referred for orthotic support if necessary, but if the child has any problems and needs to see the doctor again this is arranged. This is what would usually happen with children diagnosed with BJHS. If the participant is in the structured treatment group, they take part in a structured treatment programme. The four treatment visits include exercises and advice and take around 30 to 60 minutes each. Participants in both groups attend two more appointments with the physiotherapist 3 months and 1 year later to repeat the physical assessment and questionnaires. These appointments also take around 1 hour each time.
What are the possible benefits and risks of participating?
It is hoped that the multidisciplinary programme reduces pain and improves physical activity and participation. Improving the treatment of children with BJHS may improve not only their health but also their wellbeing and social and educational development. However, this is not guaranteed and the treatment may be of no benefit. This is the first formal study of treatment in BJHS, and the results may lead to better and more effective ways of managing this condition, raise awareness of BJHS in hospital and general practice, and help with the planning of hospital and community services to support children with this common and disabling condition. None of the treatments present a physical risk to patients, but it is possible that the exercise may make symptoms worse in some children. However, they are closely supervised and the programme of treatment is prepared by an experienced therapist and tailored to their needs. Attendance for follow up may represent a burden. However, the schedule is very similar to treatment schedules commonly used with children with many physical conditions and is generally well tolerated. Travel costs are covered.
Where is the study run from?
Norfolk and Norwich University Hospital NHS Foundation Trust (UK)
When is the study starting and how long is it expected to run for?
September 2010 to June 2014
Who is funding the study?
NIHR Research for Patient Benefit Programme (UK)
Who is the main contact?
Prof. Alexander Macgregor
a.macgregor@uea.ac.uk
Contact information
Public
Norfolk and Norwich University Hospital NHS Foundation Trust
Norwich
NR4 7UY
United Kingdom
| Phone | +44 (0)160 359 3570 |
|---|---|
| a.macgregor@uea.ac.uk |
Study information
| Study design | Randomised controlled single-site study |
|---|---|
| Primary study design | Interventional |
| Secondary study design | Randomised controlled trial |
| Study setting(s) | Hospital |
| Study type | Treatment |
| Participant information sheet | ISRCTN86573140_PIS_9-12yrs_V.4.0_18Oct2010.pdf |
| Scientific title | The efficacy and cost effectiveness of a multidisciplinary intervention strategy for the treatment of benign joint hypermobility syndrome in childhood |
| Study acronym | BENDY |
| Study hypothesis | This study aims to determine the clinical and cost effectiveness of a multidisciplinary care package in children diagnosed with benign joint hypermobility syndrome (BJH). Specifically, the aim is to conduct a clinical trial to compare a structured programme of treatment delivered through a multidisciplinary team of clinicians, physiotherapists and occupational therapists with the usual care comprising a single interview with a clinician. |
| Ethics approval(s) | Norfolk Research Ethics Committee, 23/11/2009, ref: 09/H0310/80 |
| Condition | Benign joint hypermobility syndrome |
| Intervention | Those consenting to take part will be invited for a screening assessment undertaken by the trial physiotherapist to identify their eligibility for inclusion. Written consent will be sought from eligible children and their parents. Those consenting to take part will be randomised to receive either multidisciplinary care or standard care. Minimisation will be used to ensure balance among age and gender strata. The multidisciplinary intervention will comprise an appointment with a physiotherapist for assessment and basic advice; a programme of 6 physiotherapy sessions to improve core stability and work on strengthening exercises; gait analysis and provision of foot orthoses as necessary; an OT assessment at hospital clinic and at home with provision of appliances as necessary; an OT school visit to discuss diagnosis with teachers and provide appliances as necessary. Those with problems in their hands will also receive an OT directed exercise programme. The groups will be reviewed at 3 and 12 months to assess which treatment approach was of greatest benefit in helping their pain and improving their physical functioning and quality of life. Data will also be collected on function, social activities and schooling. Data will also be collected that will allow a health economic evaluation. |
| Intervention type | Other |
| Primary outcome measure | Level of pain and physical functioning, assessed by the Childhood Health Assessment Questionnaire (CHAQ) at baseline, 3 months and 12 months |
| Secondary outcome measures | Measured at baseline, 3 months and 12 months: 1. Health-related quality of life, assessed using the CHU 9D (9 dimensional Child Health Utility) 2. Number of primary care and hospital attendances and school attendance, measured using the BJH resource use questionnaire 3. Grip strength, assessed using a hand-held dynamometer 4. Pain, assessed using the VAS/Faces Pain Score 5. Function and gross and fine motor skills, assessed using the Movement Assessment Battery for Children 6. Participation in sport, measured using a questionnaire |
| Overall study start date | 20/09/2010 |
| Overall study end date | 19/06/2014 |
Eligibility
| Participant type(s) | Patient |
|---|---|
| Age group | Child |
| Lower age limit | 5 Years |
| Upper age limit | 16 Years |
| Sex | Both |
| Target number of participants | 120 |
| Participant inclusion criteria | 1. Children aged between 5−16 years old 2. Satisfy the Brighton criteria for classification of BJS 3. Reporting musculoskeletal pain in one or more areas of the body for a duration of greater than three months 4. Written informed consent |
| Participant exclusion criteria | 1. Inflammatory joint disease 2. Identifiable heritable disorder of connective tissue 3. Presence of other chronic illness |
| Recruitment start date | 21/10/2010 |
| Recruitment end date | 31/12/2012 |
Locations
Countries of recruitment
- England
- United Kingdom
Study participating centre
United Kingdom
Sponsor information
Hospital/treatment centre
Colney
Norwich
NR4 7UY
England
United Kingdom
| Phone | +44 (0)1603 289808 |
|---|---|
| Office.R&D@nnuh.nhs.uk | |
"ROR" |
https://ror.org/01wspv808 |
Funders
Funder type
Government
No information available
Results and Publications
| Intention to publish date | 09/03/2018 |
|---|---|
| Individual participant data (IPD) Intention to share | Yes |
| IPD sharing plan summary | Available on request |
| Publication and dissemination plan | Planned publication in a high-impact peer-reviewed journal. In October 2014 a letter was posted to all participants with a document summarising the results of the study. |
| IPD sharing plan | The datasets generated during and/or analysed during the current study are/will be available upon request from Prof. Alexander Macgregor (a.macgregor@uea.ac.uk). |
Study outputs
| Output type | Details | Date created | Date added | Peer reviewed? | Patient-facing? |
|---|---|---|---|---|---|
| Participant information sheet | 18/10/2010 | 09/03/2017 | No | Yes | |
| Participant information sheet | 02/01/2009 | 09/03/2017 | No | Yes | |
| Participant information sheet | 18/10/2010 | 09/03/2017 | No | Yes | |
| Participant information sheet | version V4.0 | 18/10/2010 | 09/03/2017 | No | Yes |
| Results article | results | 01/12/2019 | Yes | No |
Additional files
- ISRCTN86573140_PIS_13-16yrs_V4.0_18Oct2010.pdf
- Uploaded 09/03/2017
- ISRCTN86573140_PIS_5-8yrs_V.1.2_Jan2009.pdf
- Uploaded 09/03/2017
- ISRCTN86573140_PIS_9-12yrs_V.4.0_18Oct2010.pdf
- Uploaded 09/03/2017
- ISRCTN86573140_PIS_Parent_V.4.0_18Oct2010.pdf
- Uploaded 09/03/2017
Editorial Notes
09/01/2019: Publication reference added.
12/07/2017: Internal edit.
