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Trial registered on ANZCTR


Registration number
ACTRN12623000249651p
Ethics application status
Not yet submitted
Date submitted
6/02/2023
Date registered
8/03/2023
Date last updated
8/03/2023
Date data sharing statement initially provided
8/03/2023
Type of registration
Prospectively registered

Titles & IDs
Public title
Running training for children with symptomatic hypermobility - a feasibility study
Scientific title
The feasibility and effect of a running training program on goal attainment in children with hypermobility spectrum disorder
Secondary ID [1] 308277 0
Nil known
Universal Trial Number (UTN)
Trial acronym
Linked study record

Health condition
Health condition(s) or problem(s) studied:
Hypermobility Spectrum Disorder 328058 0
Condition category
Condition code
Musculoskeletal 325120 325120 0 0
Other muscular and skeletal disorders

Intervention/exposure
Study type
Interventional
Description of intervention(s) / exposure
Running Intervention protocol according to Consensus on Exercise Reporting Template (CERT guidelines)

Adapted from: Gibson N, Chappell A, Blackmore AM, Morris S, Williams G, Bear N, Allison G. The effect of a running intervention on running ability and participation in children with cerebral palsy: a randomized controlled trial. Disabil Rehabil. 2018 Dec;40(25):3041-3049. doi: 10.1080/09638288.2017.1367426. Epub 2017 Aug 21. PMID: 28826274.

1. Exercise Equipment: Trampette, stairs, resistance bands, single step, cones, agility ladder, dumbbells, leg curl machine, leg press machine

2. Qualifications and training: Two or three senior physiotherapists (PTs) with experience in working with school-aged children with hypermobility spectrum disorder (HSD) will deliver the program. The lead physiotherapist has delivered this intervention for the past 8 years, undertook 15 hours training in rehabilitation of running for people with neurological conditions and undertook eight hours training in motivational interviewing which was delivered by a clinical psychologist.

3. Individual/group: Individualized programs will be established and progressed at individual rates. The individualized program will be delivered and performed in a group setting once per week and a home program provided to be performed three times per week.

4. Supervision: Participants will be supervised in a ratio of 1PT:3participants. The participants will be taught the exercises individually and once performing the exercise correctly, allowed to practice independently.

5. Adherence to exercise: Progress notes will be completed at the end of each session, including the level of difficulty of each exercise and number of repetitions, or time spent doing the exercise. Attendance will be recorded for each participant and reported as a number of sessions out of 10 possible sessions. Home program exercises will be prescribed weekly and home exercise diary sheets will be collected at the end of each week.

6. Motivation strategies: Participants will be encouraged with verbal feedback about their technique, both what was done well and what changes need to be made. Participants will be given a time or number of repetitions to aim for. Exercises will sometimes be incorporated into games. Participants will also be encouraged to “buddy up” with another group participant to encourage each other. Each participant will have a home program, with diary sheets issued and collected weekly to encourage adherence. Participants who do not attend a session without informing PTs of the reason will be called by one of the PTs the next day to encourage attendance. The PTs will use motivational interviewing techniques during the weekly one hour sessions to encourage participants to explore options for adhering to home prescribed exercises and for exploring and promoting physical activity in the community.

7a. Decision rule(s) for determining progression: Once the participant is consistently able to perform the exercise with good technique they will be progressed to the next level. Speed and quality of movement will be prioritized over load as the focus on ballistic movement is necessary for running.

7b. How program was progressed: The program incorporates a series of hierarchically challenging activities. [See Williams & Schache 2010 and Schache et al. 2014 for more detail on the activities].
Participants will be prescribed relevant activities to address the running gait impairments demonstrated for that individual. The individually tailored exercises will be derived by the therapist viewing slow motion observational sagittal and frontal video footage of the participant’s running gait and determining the abnormalities affecting the acquisition of typical running skill. The exercises/activities will target the three main muscle groups responsible for forward progression when walking and running, i.e. the ankle plantar flexors, hip flexors and hip extensors. Exercises will be progressed once good form/technique is demonstrated on the starting activity. The exercises may be performed on the trampette until the participant is able to perform the exercises overground. For simulation of leg turnover and appropriate foot contact alignment an activity termed the “claw” exercise will be utilized [See Williams & Schache 2010 and Schache et al., 2014 for more detail]. For participants whose motor control does not enable good technique, activities such as the “claw” will either be broken down into components, or facilitated with therapist handling until the participant is able to perform a cycle with good technique. This will be progressed by decreasing therapist facilitation and eventually adding resistance (for e.g., with resistance bands). Once the participant can run with good technique overground, slopes may be added, and the distance or speed increased depending on the individual goal. From slopes, participants progress to agility exercises. These began with simple cutting/side stepping exercises and progress in complexity. Once a reasonable level of agility is attained, sport specific skills relevant to the individual participant’s interest may be introduced.

8. Exercises: Please refer to Williams & Schache 2010 and Schache et al. 2014 for the types of exercises utilized to address different running impairments. Examples of exercises which may be used include: alternating feet on trampette, alternating feet with a knee lift on trampette, jumping, hopping, fast feet overground, fast feet with a knee lift, triple extension, skipping, running. Each session will utilise the following structure:
1 Warm-up: 5 min
2 Individualized exercises/activities: 45 min
3 Cool down: 5 min
4. Provision of home program: 5 min

9. Home program: All participants will receive a home program to be performed three times per week for approximately 15 minutes, which contains individually tailored exercises that have been learnt with the PTs and which they can perform independently with good technique.

10. Non-exercise components N/A

11. Type and number of adverse events: Adverse events will be recorded and followed up as per the protocol.

12. Setting: A community paediatric allied health clinic with outdoor grassed area and access to exercise equipment.

13. Exercise intervention: Participants are asked to attend one one-hour session per week with the home program performed another three times per week, for a total of 10 weeks.

14a. Generic/tailored: Each participant will receive an individually tailored program based on a core group of hierarchical exercises

14b. How the exercises are tailored: The exercises will be tailored for each participant according to the identified impairments impacting their running skill and by level of difficulty. The PTs will progress the exercises according to the participant’s response. Adjunct exercises will be added by the PTs if necessary, for example hip abductor strengthening exercises may be added if the participant cannot stabilize the pelvis while performing the exercises.

15. Decision rule for starting level: Participants will be started at the most challenging level they can perform with good technique.

16a. Adherence/fidelity: PTs will meet for 10 min following each session to discuss issues experienced by individuals in the group and find solutions.
Intervention code [1] 324732 0
Rehabilitation
Comparator / control treatment
No control group
Single case study design
Participants will be randomized to start the intervention after four, five or six weeks of baseline measures, while undertaking their usual exercise schedule.
In this design, all participants will receive the same intervention.
Control group
Active

Outcomes
Primary outcome [1] 332937 0
Completion of set goals assessed using Goal Attainment Scaling
Timepoint [1] 332937 0
T0 (start of baseline phase)
T1 (end of baseline phase)
T2 (end of 10 weeks intervention)
T3 (end of withdrawal phase, 6 weeks post-intervention completion)
Secondary outcome [1] 415204 0
Muscle Power Sprint Test
Timepoint [1] 415204 0
3-weekly throughout the study for up to 6 weeks post-intervention completion
Secondary outcome [2] 415205 0
Submaximal hopping on portable force plate to assess leg stiffness
Timepoint [2] 415205 0
3-weekly throughout the study for up to 6 weeks post-intervention completion
Secondary outcome [3] 415206 0
Adolescent Physical Activity Recall Questionnaire
Timepoint [3] 415206 0
T0 (start of baseline phase)
T3 (end of withdrawal phase, 6 weeks post-intervention completion)
Secondary outcome [4] 415207 0
KIDSCREEN-10 to assess health-related quality of life
Timepoint [4] 415207 0
T0 (start of baseline phase)
T1 (end of baseline phase)
T2 (end of 10 weeks intervention)
T3 (end of withdrawal phase, 6 weeks post-intervention completion)
T4 (follow-up, 6 months post-intervention completion)
Secondary outcome [5] 415210 0
Running gait analysis assessed by 2-dimensional video analysis of sprinting
Timepoint [5] 415210 0
T0 (start of baseline phase)
T1 (end of baseline phase)
T2 (end of 10 weeks intervention)
T3 (end of withdrawal phase, 6 weeks post-intervention completion)
T4 (follow-up, 6 months post-intervention completion)
Secondary outcome [6] 415211 0
Pain Visual Analogue Scale
Timepoint [6] 415211 0
At start and finish of each intervention session
Secondary outcome [7] 415212 0
Fatigue Visual Analogue Scale
Timepoint [7] 415212 0
At start and finish of each intervention session
Secondary outcome [8] 415213 0
Borg Rating of Perceived Exertion
Timepoint [8] 415213 0
At start and finish of each intervention session
Secondary outcome [9] 415214 0
Recruitment / Demand survey to assess feasibility
Timepoint [9] 415214 0
Baseline
Secondary outcome [10] 415215 0
Staff semi-structured interview to assess acceptability, implementation, practicality and integration, barriers and enablers to implementation of the intervention program.
Timepoint [10] 415215 0
T2 (end of 10 weeks intervention)
Secondary outcome [11] 415216 0
Semi-structured interviews with participants and carers will be assessed as a composite secondary outcome. The interviews will assess acceptability. The overarching question is “What could children with symptomatic hypermobility tell us about the value of participation in a targeted running intervention programme for their physical performance,
attendance, involvement and kinesiophobia?
Timepoint [11] 415216 0
T0 (Baseline)
T2 (end of 10 weeks intervention)
T4 (follow-up, 3 months post-intervention completion)
Secondary outcome [12] 415217 0
Attendance will be collected with study-specific attendance lists
Timepoint [12] 415217 0
Weekly during the intervention phase
Secondary outcome [13] 415218 0
Adherence to home exercise program will be assessed using a study-specific home exercise form.
Timepoint [13] 415218 0
Weekly during the intervention phase
Secondary outcome [14] 415219 0
Fidelity to the intervention protocol will be assessed using an audit of study-specific session notes.
Timepoint [14] 415219 0
Weekly during the intervention phase
Secondary outcome [15] 415220 0
Patient and carer satisfaction with the intervention as measured on a 10 point Likert scale with opportunity to comment
Timepoint [15] 415220 0
T2 (end of 10 weeks intervention)
Secondary outcome [16] 415221 0
Staff satisfaction with the intervention, as measured on a 10 point Likert scale with opportunity to comment
Timepoint [16] 415221 0
T2 (end of 10 weeks intervention)
Secondary outcome [17] 415222 0
Number and type of adverse events
Possible adverse events include falls, minor sprains and strains or asthma attack. Adverse events will be recorded on a study-specific form and the study protocol followed.
Timepoint [17] 415222 0
Weekly during the intervention phase
Secondary outcome [18] 415223 0
Evaluation of resources will be undertaken through staff semi-structured interviews.
Timepoint [18] 415223 0
T2 (end of 10 weeks intervention)
Secondary outcome [19] 415224 0
Accelerometry - wrist mounted Actigraph
Timepoint [19] 415224 0
T0 (start of baseline phase)
T3 (end of withdrawal phase, 6 weeks post-intervention completion)
Secondary outcome [20] 418186 0
Lower Extremity Grading System (LEGS) will be used to assess to assess lower extremity neuromuscular performance
Timepoint [20] 418186 0
T0 (start of baseline phase)
T1 (end of baseline phase)
T2 (end of 10 weeks intervention)
T3 (end of withdrawal phase, 6 weeks post-intervention completion)
T4 (follow-up, 6 months post-intervention completion)
Secondary outcome [21] 418187 0
Tampa Scale of Kinesiophobia
Timepoint [21] 418187 0
T0 (start of baseline phase)
T1 (end of baseline phase)
T2 (end of 10 weeks intervention)
T3 (end of withdrawal phase, 6 weeks post-intervention completion)
T4 (follow-up, 6 months post-intervention completion)
Secondary outcome [22] 418473 0
Cost of resources will be assessed using an audit of study specific financial information
Timepoint [22] 418473 0
T2 (end of 10 weeks intervention)

Eligibility
Key inclusion criteria
Children 9 to 18 years old with hypermobile Ehlers-Danlos syndrome (hEDS) or hypermobility spectrum disorder (HSD), a Beighton Score of at least 6/9, a goal to improve their running, and able to attend one training session each week for 10 weeks.
Minimum age
9 Years
Maximum age
18 Years
Sex
Both males and females
Can healthy volunteers participate?
No
Key exclusion criteria
Children will be excluded if they are unable to undertake strenuous exercise for medical reasons, have cognitive or behavioural problems that hinder working in a group, have coexisting neurological or orthopaedic conditions or have had an injury in the past 6 months that impacts their ability to run.

Study design
Purpose of the study
Treatment
Allocation to intervention
Randomised controlled trial
Procedure for enrolling a subject and allocating the treatment (allocation concealment procedures)
Methods used to generate the sequence in which subjects will be randomised (sequence generation)
Masking / blinding
Blinded (masking used)
Who is / are masked / blinded?


The people assessing the outcomes
Intervention assignment
Other
Other design features
Experimental single case-study design
Participants will be randomised to begin the intervention after four, five or six weeks of baseline measures.
Phase
Not Applicable
Type of endpoint/s
Safety/efficacy
Statistical methods / analysis

Recruitment
Recruitment status
Not yet recruiting
Date of first participant enrolment
Anticipated
1/06/2023
Actual
Date of last participant enrolment
Anticipated
1/10/2023
Actual
Date of last data collection
Anticipated
30/06/2024
Actual
Sample size
Target
8
Accrual to date
Final
Recruitment in Australia
Recruitment state(s)
WA
Recruitment hospital [1] 23521 0
Western Kids Health - Shenton Park
Recruitment postcode(s) [1] 38867 0
6008 - Shenton Park

Funding & Sponsors
Funding source category [1] 312525 0
Commercial sector/Industry
Name [1] 312525 0
Western Kids Health
Country [1] 312525 0
Australia
Funding source category [2] 312529 0
University
Name [2] 312529 0
Curtin University
Country [2] 312529 0
Australia
Primary sponsor type
Commercial sector/Industry
Name
Western Kids Health
Address
Victoria House
4 Thorburn Way
Shenton Park WA 6008
Country
Australia
Secondary sponsor category [1] 314125 0
University
Name [1] 314125 0
Curtin University
Address [1] 314125 0
Kent St, Bentley WA 6102
Country [1] 314125 0
Australia

Ethics approval
Ethics application status
Not yet submitted
Ethics committee name [1] 311860 0
Curtin University Human Research Ethics Committee
Ethics committee address [1] 311860 0
Ethics committee country [1] 311860 0
Australia
Date submitted for ethics approval [1] 311860 0
28/02/2023
Approval date [1] 311860 0
Ethics approval number [1] 311860 0

Summary
Brief summary
Trial website
Trial related presentations / publications
Public notes

Contacts
Principal investigator
Name 122626 0
Dr Annie Chappell
Address 122626 0
Western Kids Health
2/84 Matlock St.
Mt Hawthorn WA 6050
Country 122626 0
Australia
Phone 122626 0
+61 0412638093
Fax 122626 0
Email 122626 0
[email protected]
Contact person for public queries
Name 122627 0
Annie Chappell
Address 122627 0
Western Kids Health
2/84 Matlock St.
Mt Hawthorn WA 6050
Country 122627 0
Australia
Phone 122627 0
+61 8 61122977
Fax 122627 0
Email 122627 0
[email protected]
Contact person for scientific queries
Name 122628 0
Annie Chappell
Address 122628 0
Western Kids Health
Victoria House
4 Thorburn Way
Shenton Park WA 6008
Country 122628 0
Australia
Phone 122628 0
+61 8 61122977
Fax 122628 0
Email 122628 0
[email protected]

Data sharing statement
Will individual participant data (IPD) for this trial be available (including data dictionaries)?
Yes
What data in particular will be shared?
After de-identification, objective data underlying published results will be available upon request.
When will data be available (start and end dates)?
Immediately following publication, for 5 years.
Available to whom?
Data will be available to researchers who provide a methodologically sound proposal.
Available for what types of analyses?
Data will be available to achieve the aims in any approved proposal.
How or where can data be obtained?
Data will be made upon request to the primary investigator:
[email protected]


What supporting documents are/will be available?

Doc. No.TypeCitationLinkEmailOther DetailsAttachment
17567Study protocol    384892-(Uploaded-06-02-2023-13-32-57)-Study-related document.docx
17588Statistical analysis plan    Statistical Analysis Plan: Quantitative data will... [More Details]
17589Informed consent form    384892-(Uploaded-06-02-2023-13-13-35)-Study-related document.docx
17590Ethical approval    Ethical approval is being sought from Curtin Unive... [More Details]



Results publications and other study-related documents

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