Condition category
Musculoskeletal Diseases
Date applied
18/01/2017
Date assigned
09/03/2017
Last edited
12/07/2017
Prospective/Retrospective
Retrospectively registered
Overall trial status
Completed
Recruitment status
No longer recruiting

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

Trial website

Contact information

Type

Public

Primary contact

Prof Alexander Macgregor

ORCID ID

Contact details

Norfolk and Norwich University Hospital NHS Foundation Trust
Norwich
NR4 7UY
United Kingdom
+44 (0)160 359 3570
a.macgregor@uea.ac.uk

Additional identifiers

EudraCT number

ClinicalTrials.gov number

Protocol/serial number

CPMS ID: 9366

Study information

Scientific title

The efficacy and cost effectiveness of a multidisciplinary intervention strategy for the treatment of benign joint hypermobility syndrome in childhood

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

Norfolk Research Ethics Committee, 23/11/2009, ref: 09/H0310/80

Study design

Randomised controlled single-site study

Primary study design

Interventional

Secondary study design

Randomised controlled trial

Trial setting

Hospitals

Trial type

Treatment

Patient information sheet

See additional files

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

Phase

Drug names

Primary outcome measures

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 trial start date

20/09/2010

Overall trial end date

19/06/2014

Reason abandoned

Eligibility

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 type

Patient

Age group

Child

Gender

Both

Target number of participants

120

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

United Kingdom

Trial participating centre

Norfolk and Norwich University Hospital NHS Foundation Trust
NR4 7UY
United Kingdom

Sponsor information

Organisation

Norfolk and Norwich University Hospital NHS Foundation Trust

Sponsor details

Colney
Norwich
NR4 7UY
United Kingdom
+44 (0)1603 289808
Office.R&D@nnuh.nhs.uk

Sponsor type

Hospital/treatment centre

Website

Funders

Funder type

Government

Funder name

NIHR Research for Patient Benefit Programme

Alternative name(s)

Funding Body Type

Funding Body Subtype

Location

Results and Publications

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).

Intention to publish date

09/03/2018

Participant level data

Available on request

Results - basic reporting

Publication summary

Publication citations

Editorial Notes

12/07/2017: Internal edit.