Open access Protocol

Study protocol for Running for health

BMJ Open: first published as 10.1136/bmjopen-2021-057668 on 29 April 2022. Downloaded from http://bmjopen.bmj.com/ on March 17, 2024 by guest. Protected by copyright.
(Run4Health CP): a multicentre,
assessor-­blinded randomised controlled
trial of 12 weeks of two times weekly
Frame Running training versus usual
care to improve cardiovascular health
risk factors in children and youth with
cerebral palsy
Sarah E Reedman ‍ ‍,1 Leanne Sakzewski ‍ ‍,1 Lynda McNamara,2
Catherine Sherrington ‍ ‍,3 Emma Beckman,4 Kerry West,5 Stewart G Trost,4
Rachel Thomas,6 Mark D Chatfield,1 Iain Dutia,4 Alix Gennen,1 Bridget Dodds,1
Zoë Cotton,1 Roslyn N Boyd1

To cite: Reedman SE, ABSTRACT

Sakzewski L, McNamara L, et al. Strengths and limitations of this study
Introduction Children and youth with moderate-­severe
Study protocol for Running (Gross Motor Function Classification System (GMFCS)
for health (Run4Health CP): a ► This randomised controlled trial of Frame Running
levels II–V) cerebral palsy (CP) participate less frequently
multicentre, assessor-­blinded training in children and youth with cerebral palsy is
in physical activities compared with peers without CP
randomised controlled trial powered to detect change on the primary outcome
of 12 weeks of two times and have elevated risk of cardiorespiratory morbidity and measure of cardiovascular fitness.
weekly Frame Running training mortality in adulthood. Frame Running (RaceRunning) is ► Retention (sustainability) of changes will be exam-
versus usual care to improve a new athletics discipline that is an accessible option for ined at a follow-­up 12 weeks after the training ses-
cardiovascular health risk physical activity participation for people with moderate-­ sions are complete.
factors in children and youth severe CP. There is no high-­quality evidence for the effect ► Children and youth with severe functional mobility
with cerebral palsy. BMJ Open of Frame Running on cardiovascular disease in children
2022;12:e057668. doi:10.1136/ limitations and intellectual disability will be included.
and young people with CP. The primary aim of this study ► A validated maximal exercise test will not be
bmjopen-2021-057668
is to conduct a randomised controlled trial of the effect conducted.
► Prepublication history for of 12 weeks of Frame Running training on risk factors for
this paper is available online. cardiovascular disease.
To view these files, please visit Methods and nalysis Sixty-­two children and youth with
the journal online (http://dx.doi.​ CP (age 8–20 years) in GMFCS levels II–V will be recruited of Queensland Human Research Ethics Committees have
org/10.1136/bmjopen-2021-​ approved this study. Results will be disseminated in peer-­
across four sites and randomised to receive either 12
057668).
weeks of Frame Running training two times weekly for reviewed journals and scientific conferences; through
Received 23 September 2021 60 min, or usual care. Outcomes will be measured at professional and athletic organisations; and to people with
Accepted 22 March 2022 baseline, immediately postintervention (primary endpoint) CP and their families.
and 12 weeks later for retention of training effects. The Trial registration number ACTRN12621000317897;
primary outcome is cardiorespiratory fitness as measured Australian New Zealand Clinical Trials Registry number.
© Author(s) (or their by distance covered on Six Minute RaceRunner Test with
employer(s)) 2022. Re-­use 1 min heart rate recovery. Other outcomes include blood
INTRODUCTION
permitted under CC BY-­NC. No pressure, objectively measured physical activity, body
commercial re-­use. See rights One in 700 Australians has cerebral palsy
mass index, waist circumference, percentage body fat,
and permissions. Published by
gross motor function capacity, community participation,
(CP), a permanent but not unchanging
BMJ. disorder of posture and movement caused
feasibility, tolerability and safety. Adverse events will be
For numbered affiliations see
monitored, and participants and their caregivers will be by a disturbance to the developing fetal or
end of article.
interviewed to discern their experiences of participation in infant brain.1 Children with CP participate
Correspondence to Frame Running. in physical activities less often compared with
Dr Sarah E Reedman; Ethics and dissemination The Children’s Health peers without CP.2 Children with CP also have
​s.​reedman@​uq.​edu.​au Queensland Hospital and Health Service and the University high levels of sedentary behaviour,3 apparent

Reedman SE, et al. BMJ Open 2022;12:e057668. doi:10.1136/bmjopen-2021-057668 1

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from early infancy and peaking by 4 to 5 years of age when

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followed through to middle childhood.4 Adults with CP
experience increased risk of non-­communicable diseases
associated with low physical activity, including cardio-
vascular disease, mental illness, osteoporosis and osteo-
arthritis (Odds Ratios 1.3–5.8).5 There is evidence that
the disparity in non-­communicable disease risk begins
early, with a large population-­based cohort study demon-
strating increased risk of mental health disorders in chil-
dren (6–17 years of age) with CP compared with children
without CP.6 In this study, pain and low physical activity
level explained part of the relationship between CP and
depression.6
Life expectancy in people with CP in general is only
slightly reduced compared with the people without CP, Figure 1 Petra Frame Runner(TM) by ConnieHansen
however, those individuals with moderate-­severe motor running frame.
impairments have significantly lower life expectancy.7
The causes of early death in people with CP are most
frequently respiratory and cardiovascular diseases,8 with severe mobility limitations (see figure 1). In the absence
respiratory illness the leading cause of death in children of an existing systematic review of the literature, an author
with CP.9 An Australian prospective population-­ based search was conducted on 21 September 2021 for articles
register study following n=3507 individuals with CP deter- indexed in the PubMed database, using the terms ‘Race-
mined that inability to walk independently (an indicator Running’ OR ‘Frame Running’ OR ‘race running’ AND
of severe CP) was the strongest predictor of mortality in ‘cerebral palsy’ located in title/abstract (with additional
people with CP (adjusted Hazard Ratio 6.2).10 There is hand search of reference lists for included articles). This
expert consensus that increasing aerobic fitness and phys- search returned only seven studies: one pilot single-­group
ical activity in children with severe CP is likely to amelio- pre–post trial,14 one study protocol for a pilot randomised
rate the severity of acute respiratory illness.9 Despite this, feasibility study,15 one reliability study for a Frame
recent systematic reviews have demonstrated that there Running-­specific field exercise test,16 two cross-­sectional
are no effective physical activity interventions for people studies examining relationship between impairments and
with CP that do not walk independently, and interven- Frame Running performance17 18 and two cross-­sectional
tions for children who can walk independently may not studies on kinesiologic and metabolic responses or adap-
have a clinically meaningful effect on physical activity tations to use of running frames.19 20 The pre–post pilot
behaviours, including habitual physical activity and partic- trial included n=15 adolescents and young adults with CP
ipation in physical activities.11 12 Contributing factors to (age range 9–29 years, Gross Motor Function Classifica-
inefficacy may have included: selection bias (inclusion tion System (GMFCS) levels I–IV) and demonstrated that
of children with the highest level of physical activity 12 weeks of two times weekly Frame Running training
and physical functioning), failure to address environ- led to on average, a 34% increase in cardiorespiratory
mental, contextual and behavioural barriers to physical endurance and a 9% increase in thickness of the medial
activity, issues with outcome measurement11 and dosing gastrocnemius muscle.14 Frame Running can evoke a
below minimum recommended guidelines.13 It is clear heart rate commensurate with high-­intensity exercise19
that there is an urgent need for high-­quality research and uses large muscle groups in a reciprocal way that
into physical activity interventions of sufficient dose and may have functional cross over to enhanced mobility.20 A
duration in youth with CP who have major limitations in larger (n=25) pre–post pilot study of once weekly Frame
walking ability. Furthermore, such interventions need to Running training for 24 weeks duration is planned.15 This
be safe, community-­based, informed by consumer needs, study with no accompanying sample size calculation has
and aimed to enable ongoing, normal community partic- the potential to be underpowered and/or underdosed
ipation and inclusion. to detect improvements in cardiometabolic risk factors.
Frame Running, formerly known as RaceRunning, is Furthermore, as the study is unrandomised, the quality
a para-­athletics discipline recently sanctioned by World and certainty of the evidence provided will necessarily be
Para Athletics (the International Paralympic Sports lower than a randomised study.
Federation for athletics). Frame Running was invented We, therefore, aim to conduct an adequately powered
in 1991 by Connie Hansen, an Occupational Therapist randomised controlled trial of Frame Running training
and para-­ athlete and Mansoor Siddiqi, a para-­ athlete in children and youth with CP on cardiometabolic risk
with CP competing in the now defunct discipline of back- factors and related outcomes (Run4Health CP). This
ward wheelchair racing (foot-­propelled). Frame Running study may, therefore, provide evidence that cardiometa-
uses a three-­wheeled frame with low rolling resistance bolic risk factors can be modified in children and youth
for support, enabling running in people with otherwise with CP who have moderate to severe motor impairment

2 Reedman SE, et al. BMJ Open 2022;12:e057668. doi:10.1136/bmjopen-2021-057668

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and high support needs in mobility. This evidence may more than six sessions of formal Frame Running training

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have critical patient and clinical impacts through support with a coach or health professional within the last 6
of funding for running frames and may help to foster months, (5) can understand and follow the directions of
development of the discipline and expand participation the coach and assessors for the purposes of training safely
opportunities. and completing outcome measurement in the opinion of
the principal investigator.
Participants are excluded if at any time: (1) the child/
METHODS AND ANALYSIS youth has orthopaedic and/or neurological surgery within
Objectives 6 months prior to baseline or during the study period
The primary objective of this study is to compare the requiring a period of recovery that would exclude the
effect of 12 weeks of Frame Running training versus usual participant from training for more than 1 week, (2) the
care control on cardiovascular fitness (endurance) on the child/youth has uncontrolled epilepsy, medical fragility
Six Minute RaceRunner Test (6MRRT) and 1 min heart and/or serious precautions not able to be accommodated
rate recovery (HRR1min) following exercise testing imme- (eg, significant history of atraumatic lower limb fractures
diately at postintervention (primary endpoint) and at 12 or sacral pressure injuries, etc) precluding participation
weeks postintervention. in moderate-­vigorous intensity Frame Running, (3) care-
Secondary objectives are to compare the effect of 12 giver English language skills are not sufficient to under-
weeks of Frame Running training versus usual care stand the study information, provide informed consent
control immediately postintervention and at 12 weeks and/or complete study questionnaires.
postintervention on:
1. other cardiovascular risk factors including: resting Interventions
blood pressure (BP), habitual physical activity lev- Frame Running training group
el, body mass index, per cent body fat and waist Frame Running training will consist of two, 60 min sessions
circumference. per week for 12 consecutive weeks (total dose 24 hours).
2. Gross motor activity capacity including gross motor Established guidelines for aerobic exercise to improve
function and Frame Running-­specific activity limita- cardiovascular health in typically developing individuals
tion tests. recommend a minimum frequency of three sessions per
3. Community participation. week.13 There is evidence, however, that two sessions per
The tertiary objective of this study is to determine week is adequate in deconditioned individuals with CP
whether 12 weeks of Frame Running training is feasible, to improve aerobic fitness,13 and this was demonstrated
tolerable, safe and sustainable in the study population, in the pilot pre–post study of Frame Running training by
including whether participants report that it induces Hjalmarsson et al.14 This can likely be attributed to the
additional pain and fatigue when compared with usual dose–response relationship between physical activity and
care. cardiorespiratory outcomes, whereby even small increases
in physical activity in previously inactive individuals can
Trial design result in clinically meaningful improvements in health.21
Run4Health CP is a pragmatic, single (assessor)-­ blind Provision of only two sessions per week may also increase
randomised controlled, multicentre trial with two parallel the likelihood that participants can comply with the inter-
groups. The primary timepoint is immediately postin- vention (considering issues such as time and financial
tervention (12 weeks postbaseline) and the secondary constraints relative to a third session).
timepoint is 12 weeks postintervention (24 weeks post- Participants will attend Frame Running training in
baseline). The study will be conducted in four Australian groups of approximately three, matched by age and/or
cities, Brisbane (n=24), Cairns (n=18), Sydney (n=10) ability if possible. Sessions will be administered by a coach
and Sunshine Coast (n=10). Assessment of outcome with qualifications in Physiotherapy and/or Exercise Phys-
measures and Frame Running training will be conducted iology. Participants in the training group are permitted to
at community synthetic athletic tracks and nearby associ- receive their usual care from non-­study providers (as per
ated indoor sports facilities at a time convenient to partic- concomitant care) with no restriction.
ipants and their caregivers. Recruitment commenced on Frame Running training sessions will consist of a
16 August 2021 and the first participant was enrolled on combination of (1) anaerobic Frame Running (ie, starts
16 September 2021. Last participant data collection is and sprints drills using established athletic training prin-
anticipated in January 2023. ciples), (2) aerobic Frame Running (ie, steady running
working towards ≥15 min duration) and (3) task-­specific
Eligibility criteria functional training for Frame Running technique
Participants eligible for the trial must comply with all and skills (eg, braking, steering, propulsion strategies,
of the following eligibility criteria at randomisation: running form and power). Training sessions will increase
(1) diagnosis of CP and classified in GMFCS levels II–V, in difficulty in a stepwise fashion, with the aim to initially
(2) between 8.00 and 20.99 years of age, (3) live within develop basic skills in operating a running frame, working
150 km of one of the trial sites, (4) have not engaged in towards maintaining moderate-­vigorous exercise intensity

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throughout a 60 min session. A training load of 60%–75% Following outcome measurement at the final time point,

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of peak heart rate can elicit a 9%–40% increase in peak participants in the control group will be provided with
aerobic capacity with 2–4 sessions per week for minimum an information package regarding local Frame Running
20 mins in individuals with CP.13 Based on a literature training sessions, running frame fitting results (eg, frame
review of exercise training studies, proposed ideal exer- size required and attachments) and advice about how to
cise parameters for individuals with CP are: an intensity obtain a running frame and participate in the sport. They
between 60%and 95% of peak heart rate, between 40% will also receive up to two phone calls from a therapist
and 80% of the heart rate reserve or between 50% and providing physical activity counselling and advice. The
65% of VO2peak.13 To monitor adherence to this exercise aim of this package of supports is to improve equity in
intensity, participants will wear a Polar Verity Sense (Polar access to Frame Running opportunities for those other-
Electro Oy, Kempele Finland) optical heart rate monitor wise receiving a no-­treatment control.
on the non-­dominant upper limb during training sessions
with output observed by the coach and/or assistants (eg, Modifications and adaptations
undergraduate physiotherapy or exercise physiology Heart rate data will be used to adjust the session in real
students). As suggested by Verschuren et al, peak heart time and to tailor the progression of session difficulty
rate will be estimated at 194 bpm for children and youth from week to week, so that the participant spends at least
with CP in the absence of maximal exercise testing.22 60% of session duration in the target HR range. Individual
Therefore, the target heart rate will be between 116 bpm tailoring will also accommodate variability in participants’
and 185 bpm. Where possible, when a participant provides propulsion strategies, motor type/distribution, activity
a Global Positioning System (GPS)-­enabled smartphone, limitations, age and interests. If any unexpected, unusual
distance covered and time in motion will be recorded or additional pain or fatigue beyond what is considered
by attachment of the smartphone to the running frame ‘normal’ is experienced by a participant allocated to the
using the Polar Beat phone application (Polar Electro Oy, Frame Running training group, this will be discussed with
Kempele Finland). the participant and their caregiver and modifications to
Running frames are registered in the low-­risk Medical the training programme may be implemented. Unex-
Device Class 1 category on the Australian Register of pected or unusual pain or fatigue in the training group
Therapeutic Goods. They are manufactured overseas and will be recorded as an adverse event. Other participant
are imported to Australia by Dejay Medical and Scientific. characteristics may necessitate modification or adapta-
There are currently two brands available in Australia, tion to the training programme, including but not limited
‘RAD—Trike—Disability vehicle, cycle, tricycle, foot-­ to intellectual disability, injury, hearing and/or vision
propelled’ (ARTG: 345236) and ‘By Connie Hansen— impairment, tactile and/or proprioceptive impairment,
Disability vehicle, cycle, tricycle, foot-­propelled’ (ARTG: behavioural and/or emotional dysregulation. Modifica-
309224). Both types of running frames may be used in the tions may include reduction in dose, changes to training
trial according to availability and suitability, as the differ- session content, use of assistive technology (hearing aids,
ences between these brands are expected to be superficial visual aids, etc), visual guides, caregiver involvement and/
considering the context of the trial (novice and beginner or advice and education regarding management of pain,
Frame Running athletes, recreational style participation injury and fatigue.
with elementary competition). Where possible, the same
frame will be used by each participant throughout the Adherence and fidelity
study period with consistent attachments, seat height The training content has been manualised to facilitate
and chest plate angle/depth unless these are adapted for consistent application by coaches across trial sites and
performance reasons. participants and to promote adherence to the prescribed
dose. Several strategies will be applied to optimise the
Usual care control group participant’s frequency of attendance at sessions and
Participants in the control group will receive their usual level of involvement (which is defined as the subjective
care from non-­ study providers (type/dose/content as experience of participation while attending and includes
per concomitant care). This will determine the effect of elements such as engagement, motivation, persistence and
Frame Running training in addition to usual care, which affect).23 These strategies are hypothesised to fulfil partic-
already contains active treatments such a physiotherapy ipants’ basic psychological needs of autonomy, compe-
and/or occupational therapy. Participants in the control tence and relatedness according to self-­ determination
group will not be offered Frame Running training and will theory,24 which has been demonstrated to underpin phys-
be asked to refrain from participating in Frame Running ical activity interventions in children with CP25:
until they have exited the study, however, this will not be 1. Training activities will be individually tailored as de-
actively prevented for ethical reasons. As Frame Running scribed above. This is likely to facilitate a ‘just right chal-
requires access to a running frame, and participants are lenge’ and fulfil participants' need for competence.25
not expected to have their own frame, it is expected that 2. Training will occur in groups of approximately three,
few participants in the control group will participate in matched where possible for age and/or ability. Train-
Frame Running during the study period. ing together with peers, with social group dynamics

4 Reedman SE, et al. BMJ Open 2022;12:e057668. doi:10.1136/bmjopen-2021-057668

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managed carefully, is likely to promote social connec- exercise test, however, it is likely that many participants

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tion and fulfil participants’ need for relatedness.26 will achieve almost maximal heart rate.
3. Coaches will use autonomy-­ supportive, empathet-
ic communication with participants and families. Secondary outcomes
Coaches will facilitate participant self-­efficacy through Cardiovascular health (secondary)
teaching positive self-­talk about performance and will HRR1min will be taken immediately following the 6MRRT.
promote positive peer-­to-­peer encouragement.25 HRR1min in beats per minute is the difference between
Furthermore, training sessions will take place in loca- the heart rate taken at the cessation of the 6MRRT and
tions where Frame Running squads train on a regular exactly 60 s later, while the participant is engaged in rela-
basis. This provides an ongoing avenue for normal, tive rest (ie, has stopped moving).27 HRR1min is strongly
regular participation in the sport once the free clinical associated with cardiac mortality and is responsive to a
trial sessions conclude. 12-­week cardiac rehabilitation programme in children
Individual adherence to the training manual will following heart surgery.27 Children and youth will wear a
be recorded on a session-­ by-­
session basis by coaches. Polar Verity Sense Optical heart rate monitor on the less-­
Measures include: impaired upper arm during testing.
1. Percentage of sessions attended (including partial Resting BP will be measured using an automated arm-­
attendance). cuff sphygmomanometer (valid and reliable).28 Resting
2. Percentage of training drills completed according to systolic and diastolic BP in mm Hg is a traditional risk
the manualised content. factor for cardiometabolic disease in individuals with
3. Percentage of session duration spent within the target CP,28 and systolic BP is associated with cardiorespiratory
heart rate threshold for training intensity. fitness, central adiposity and body mass index (BMI) in
4. List of modifications or adaptations. children with CP.29 Systolic and diastolic BP were respon-
5. Distance covered and time in motion during the train- sive to a 12-­week training programme in youth with Down
ing session. syndrome.30
Reasons for missed or incomplete sessions will be Habitual physical activity will be quantified using accel-
recorded. Adherence data will be reported alongside erometry, a valid, reliable and feasible method in youth
study outcomes. with moderate to severe CP.31 Participants will wear an
ActiGraph GT3X+ on the less-­ impaired wrist and less-­
Concomitant care impaired anterior thigh for 7 days during waking hours
Participants in both groups may continue any usual care during their usual activities (free-­ living). Data will be
from non-­study providers throughout the study period processed to identify time spent in different postures and
(except Frame Running in the usual care control group). activities using machine learning algorithms; a combined
Type, dose and duration of usual care are likely to vary thigh and wrist classification model has been validated in
widely between participants owing to individual needs, children classified in GMFCS levels III and IV.31
access and funding arrangements. This could include BMI, kg/m2 will calculated according to the equa-
Botulinum Toxin-­A injections, serial casting and a broad tion: BMI=weight (Kg)/height2 (m). Weight will be
array of exercise and movement-­based therapies. Partic- taken using the same calibrated digital scale at each site
ipants in both groups will be asked to record frequency and height taken using the same stadiometer at each
of participation in Frame Running and other physical site for all participants. Participants who are unable to
activities, and frequency/type/dose of usual care ther- stand unassisted will access chair scales. If body shape
apies from non-­study providers from allocation to exit distortion is severe and/or standing height is not
using a Usual Care Diary. Botulinum Toxin-­A injections feasible, then height will be measured using a recum-
and serial casting are not expected to have significant bent measuring board if available or will be estimated
impacts on our activity and participation-­level outcome using segmental limb length (knee height).32 Anthro-
measures at the group level. Based on prior experience in pometric measures will be converted to Z-­scores using
randomised controlled trials administered by our centre, age and gender-­specific reference data for the general
it is not feasible to exclude participants receiving these population.33
interventions as timely recruitment would be affected. Waist circumference (cm) will be measured to the
nearest millimetre at the midline level (midway between
Outcomes the superior border of the iliac crest and the inferior rib
Primary outcome margin, often slightly above the umbilicus) using a non-­
Cardiovascular health (primary) stretchable tape measure.34
Distance (metres) covered in the 6MRRT.16 The 6MRRT Percentage body fat will be estimated based on the
is a validated measure of RaceRunning endurance with triceps and subscapular skinfold thickness using CP-­spe-
good test–retest reliability (Intraclass Correlation Coeffi- cific equations.35 This will be measured using callipers
cient (ICC)=0.78–0.91) in children classified in GMFCS (Harpenden Skinfold Caliper, Baty International, West
levels III and IV. The 6MRRT is theoretically a submaximal Sussex UK) by trained investigators.

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Gross motor capacity income, residential postal code, presence of comorbid

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Gross motor function will be assessed using GMFM-­66, diagnoses, list of up to nine sports/PAs the participant
a criterion referenced observation measure developed attended in the last 12 months and caregiver frequency
using Rasch modelling to measure gross motor function of participation in structured and unstructured sports/
of children with CP.36 The GMFM-­ 66 has established PAs in the last 4 months. Participants will also be screened
construct validity, high test–retest reliability (ICC 0.99) for medical conditions that may be precautions to high-­
and is responsive to change.36 37 intensity exercise using a running frame requiring atten-
Frame Running-­ specific activity limitation will be tion or adaptation but not meeting exclusion criteria (eg,
assessed using 100 m sprint (time in seconds), distance known stable cardiovascular or respiratory condition,
covered in four strides (metres, average of best two trials etc.).
of three), and step count in 20 m (steps, average of best
two trials of three). The assessment of function is activity Participant tmeline
specific and, therefore, outcomes should be strongly Run4Health CP schedule of assessments and inter-
related to the activity of interest.38 These assessments are ventions are provided in table 1 and the Consolidated
investigator developed and will be subjected to further Standards of Reporting Trials (CONSORT)52 study flow
independent assessment of their validity and reliability. diagram is provided in figure 2.

Participation
Sample size
Community participation will be evaluated using the
Based on the primary outcome of 6MRRT, which has a
Participation and Environment Measure for Children
smallest detectable difference of approximately 150 m
and Youth (PEM-­ CY).39 The PEM-­ CY is a caregiver-­
and sample SD of 150 m,16 a sample size of n=44 will
report questionnaire with good test–retest reliability and
detect at least this difference at 90% power and two-­sided
internal consistency.39 Youth 18 years and older will be
5% significance level. To allow for up to 15% attrition,
invited to self-­report the questionnaire. Summary scores
n=52 (n=26 per group) will be required, however, addi-
for participation frequency, involvement and per cent
tional funding awarded to increase the implementation
environmental supportiveness will be calculated.
of Frame Running in a fourth site (Sunshine Coast) will
allow for up to n=62 participants.
Feasibility, tolerability and safety
Feasibility, tolerability and safety will be measured on a
weekly basis in both groups using the Wong-­Baker FACES Recruitment
rating scale (pain),40 Fatigue Severity Scale (fatigue),41 Strategies to achieve adequate participant enrolment to
and for the training group only, training load (Rate of reach the target sample size are as follows:
Perceived Exertion (RPE) on the OMNI RPE42 multi- 1. Clinical database: potential participants will be identi-
plied by session duration).43 Monitoring of adverse and fied on a clinical database held and maintained by the
unintended events including injuries will be undertaken Queensland Paediatric Rehabilitation Service (QPRS)
throughout the study. at the Queensland Children’s Hospital and the Sydney
Children’s Hospitals Network (SCHN). Caregivers of
Classification systems and demographic characteristics children/youth who have previously consented to re-
The following validated classification systems will be ceive communications about research studies will be
applied: GMFCS Expanded and Revised,44 Manual Abil- sent a copy of the study flyer to their contact email or
ities Classification System,45 Communication Function postal address.
Classification System,46 Visual Function Classification 2. Clinical service: children and youth with CP attending
System (VFCS),47 Eating and Drinking Ability Classifica- an associated clinical service within QPRS and SCHN
tion System (EDACS).48 The VFCS and EDACS will be will be identified by their treating clinicians based on
applied owing to the contribution of the visual system eligibility criteria. Clinicians will ask permission to dis-
to athletic performance,49 and the association between cuss the project and gain consent from the family to be
eating and drinking ability and nutrition status, which contacted by a project staff member.
is relevant to body composition, muscle mass, func- 3. Patient advertising: patient waiting areas at associated
tional ability and performance in training programmes clinical services within QPRS and SCHN will display
in people with CP.50 51 If known, the participant’s Frame the approved flyer during the recruitment period.
Running Sport Class under the two existing classification 4. Newsletter: the flyer will be included in the newsletters
systems will be recorded (RR1/RR2/RR3 and/or T71/ distributed by associated clinical services within QPRS
T72). If unknown (or not yet classified), a provisional and SCHN and research groups of the investigators.
classification will be performed following the process 5. Websites: the flyer will be posted on the research web-
outlined by Athletics Australia. sites of the investigators.
The following participant demographic characteris- 6. Social media/word of mouth: the flyer will be posted
tics will be collected to characterise the sample: partici- on social media websites, which may include but are
pant age, sex, dominant hand, self-­reported household not limited to Facebook, Twitter and Instagram. The

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Table 1 Schedule of assessments for Run4Health CP study
Immediately post-­ Retention (24
Timepoint Enrolment Allocation/baseline Intervention intervention (12 weeks) weeks)
Visit number: Screen T1   T2 T3
Participant contact X        
Eligibility screen X        
Informed consent X        
Allocation   X      
Interventions:          
Frame running training     X    
Usual care control     X    
Assessments:          
Classification systems (GMFCS, MACS,   X      
CFCS, VFCS, EDACS)
Demographic questionnaire   X      
Frame Running provisional sport class   X      
Six minute RaceRunner test   X   X X
Heart rate recovery in 1 min   X   X X
Resting blood pressure   X   X X
7 day free-­living accelerometry   X   X X
for habitual physical activity
(ActiGraphGT3X+thigh and wrist)
Body mass index   X   X X
Per cent body fat   X   X X
Waist circumference   X   X X
Gross Motor Function Measure 66   X   X X
Frame Running activity limitation tests   X   X X
Participation and Environment Measure   X   X X
for Children and Youth
Wong-­Baker FACES rating   X X X X
Fatigue severity scale   X X X X
Monitoring of adverse and unintended   X X X X
events
Usual care diary   X X X X

CFCS, Communication Function Classification System; EDACS, Eating and Drinking Ability Classification System; GMFCS, Gross Motor Function
Classification System; MACS, Manual Abilities Classification System; VFCS, Visual Function Classification System.

electronic version of the flyer may then be shared by not blind to treatment allocation, procedures for emer-
third parties. gency unblinding are not required.

Allocation and blinding (masking) Data collection

Participants will be randomly assigned to either Frame Assessment order
Running training or usual care control with a 1:1 alloca- Assessments will be delivered in a standardised order
tion as per a computer-­generated randomisation schedule across two sessions (first lab-­based, second track-­based)
using the Research Electronic Data Capture (REDCap) on different days to reduce the effect of fatigue and
randomisation module, stratified by GMFCS (II–III/ enable a familiarisation session with the running frame
IV–V) and site (Brisbane vs Cairns vs Sydney vs Sunshine prior to track-­based assessments. The track-­based assess-
Coast), using permuted blocks of random sizes. Randomi- ments will be delivered in the following order: 100 m
sation will occur following enrolment into the study and sprint, distance in four strides, step count in 20 m and
completion of all baseline assessments except for 7-­day 6MRRT with adequate rest in between.
habitual PA monitoring. Table 2 contains information
about concealment and blinding (masking), who these Interventionist training and experience
apply to, how and when. As participant health and safety The interventionists (Frame Running coaches) will be
is managed directly by Frame Running coaches who are exercise physiologists, physiotherapists and/or athletics

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measures. Regular supervision meetings will be conducted

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to facilitate adherence to the assessment manual.

Retention
Participant retention
The following strategies will be used to promote partic-
ipant retention and complete follow-­ up: (1) Frame
Running training will be offered at no cost, (2) where
possible according to track availability, sessions will be
scheduled at mutually convenient times, (3) question-
naires will be administered using the REDCap survey
module enabling forced choice/completion and auto-
mated email reminders, (4) usual care control partic-
ipants and/or their caregivers will be reminded that a
Frame Running participation pack with interventionist
follow-­up support will be provided after the T3 retention
(24 weeks) timepoint is complete, (5) once enrolled,
investigators will make all reasonable efforts (including
phone call, email and text messages) to contact partici-
pants and/or their caregivers to encourage completion
of overdue assessments, if any.

Participant withdrawal
Participants can withdraw at any time. Participants who
choose to withdraw from the study will not be penalised
in any way. They will be assisted to source another local
therapy option that matches their preferences if desired.
Figure 2 Consolidated Standards of Reporting Trials Participants are informed of their right to withdraw at any
(CONSORT) study flowchart. GMFCS, Gross Motor Function time without consequences at the time of reading partic-
Classification System.
ipant information forms and signing of consent forms.
Any deidentified (including reidentifiable) data collected
from participants who later withdraw will be retained and
coaches with at least 2 years’ experience prescribing
included in analyses. Reasons for participant withdrawal
physical activity programmes to people with disabilities
will be recorded and reported where available.
including CP and conducting group exercise sessions
with children and young people. Interventionists will Data management and access
have current cardiopulmonary resuscitation and first aid Study data will be collected and managed using REDCap
qualifications and will adhere to institutional policies and electronic data capture tools hosted at The University
procedures for child safety. of Queensland.53 54 REDCap is a secure, web-­ based
Interventionists will be provided with 6 hours face-­to-­ software platform designed to support data capture for
face didactic training from the principal investigator in research studies. To promote data quality and mini-
how to deliver the intervention according to the training mise data loss, REDCap forms will be set up with range
manual. The following topics will be covered: (1) general checks and forced completion. All assessments adminis-
principles of aerobic and anaerobic exercise in CP, (2) tered by the outcome assessors for backup if recording
coaching principles to provide a fun and intrinsically forms are incomplete, damaged or lost. The University
motivating exercise experience, (3) interpreting and of Queensland Research Data Manager database will
applying the Frame Running intervention manual, (4) be used for long-­term data storage, and a description
correctly fitting athletes to running frames and (5) prac- of the data will be uploaded onto the UQeSpace repos-
tical component. Regular supervision meetings will be itory at the conclusion of data collection and analysis.
conducted throughout the trial to facilitate adherence to Confidentiality of participant data will be maintained
the training manual. at all times from collection to storage. A deidentified
data set will be made available on written request for
Outcome assessor training and experience the purposes of further scientific research, including
Outcome assessors will be physiotherapists with at least meta-­analysis, ancillary studies related to the original
3 years’ experience administering the GMFM-­66 to chil- aims and objectives and verification of results.
dren and youth with CP and will have completed the
GMFM Criterion Test for scoring reliability. They will be Statistical methods
provided with written and videotaped standardised proce- Between-­ group differences for primary and secondary
dures for the administration of all other study outcome outcomes will be determined on an intention-­ to-­
treat

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Table 2 Blinding (masking) and concealment information for the Run4Health CP trial
Group or individual blinded Information withheld Method of blinding
Person assigning participants to groups Group assignment (REDCap) randomisation module, with schedule
generated and entered by a biostatistician not
otherwise involved in participant recruitment,
assessment or trial conduct. Trial staff with access to
the randomisation function (to allocate participants)
do not have access to the randomization schedule.
Participants Not blinded after baseline  
Coaches delivering intervention Not blinded after baseline  
Outcome assessors Group assignment Not told of group assignment and no access to
randomisation status or intervention information on
REDCap. Participants and caregivers will be asked
not to discuss their assignment with the outcome
assessor. Questionnaires will be entered directly into
REDCap by participants and/or their caregiver and
will be locked for editing by study personnel except
for one research data manager (who is not on the
investigator team) if an error in data entry is made. All
changes to data are available in a log accessible from
REDCap.
Research data manager/study Not blinded after baseline  
coordinator
Statistician Group identity The analysis code is written and finalised before the
dataset is made available for analysis. The groups
are randomly assigned as ‘group A’ or ‘group B’ in
the downloaded dataset provided to the statistician.
The identity of the group is revealed after the primary
statistical analysis is complete.
Investigators and manuscript writers Not blinded  
CP, cerebral palsy.

basis using generalised estimating equations to account wearing accelerometers, with a focus on preventing
for the repeated measures design, stratification and the development of pressure areas, early identifica-
potential missing outcome data.55 Covariables will be tion of allergic skin reactions and reducing unpleasant
stratification factors (GMFCS II–III vs IV–V and site), sensations, (3) treating/assessing staff will be provided
baseline and wear time for accelerometer data that may with standardised training that includes a component
be confounded by duration of wear, for example, average on awareness of risks, application of control strategies
minutes per day of sedentary behaviour. Effect estimates and safety, (4) participants may use running frames
will be presented as a mean difference and 95% CI with only with a properly fitting, Australian-­ standards
a significance level of p<0.05. Data will be inspected visu- approved bicycle helmet and appropriate footwear of
ally for normality, homoscedasticity and linearity. If any their choice, including orthoses if preferred (same
analyses are found to violate necessary assumptions, then footwear to be worn for all assessments), (5) partici-
data will be transformed, or appropriate non-­parametric pants will be reminded to use sun protection and have
analysis methods will be used. access to fresh drinking water during training sessions,
Data monitoring and safety (6) participants will be instructed on safe use of the
There are no additional risks to participating in Frame running frame at a familiarisation session of at least
Running beyond typical physical activity participation 30 min, (7) participants will be encouraged to wear
using adaptive equipment in this population. The padded bike pants to reduce discomfort in the saddle
following control strategies will be implemented to and (8) fatigue and pain will be monitored on an
manage the risk of adverse events: (1) participants ongoing basis and training load adapted accordingly.
will be screened for the presence of comorbid condi- Coaches and assessors are asked to report adverse
tions and will be managed by senior experienced events in real time using REDCap, which is monitored
clinical staff including the principal investigator, (2) by the principal investigator, who will determine the
families will be provided with an information sheet severity of the adverse event, whether it is expected
and brief counselling on the risks associated with or unexpected, and whether it is related or unrelated

Reedman SE, et al. BMJ Open 2022;12:e057668. doi:10.1136/bmjopen-2021-057668 9

Open access

to the intervention. Serious or unexpected adverse Ethics and dissemination

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events will be discussed at the earliest convenience by Run4Health CP is registered on the Australian New
the chief investigators (SER, LS, LM, CS and RNB) and Zealand Clinical Trials Registry. Protocol updates will
reported to the Ethics committees, at which point a be reflected in the trial registration and reported in the
decision will be made about continuing the trial. No primary result manuscript. The project has received
interim analyses will take place. The study is covered by ethics approval from the Children’s Health Queensland
standard clinical trials insurance held by The Univer- Hospital and Health Service Human Research Ethics
sity of Queensland. Committee (HREC/21/QCHQ/69281) and the
University of Queensland Human Research Ethics
Qualitative interviews Committee (2021/HE000725). Results of the study will
To fully address the tertiary objective of this study, semi- be published/disseminated in (1) the trial registration
structured interviews will be conducted in up to eight database, (2) conference abstracts and presentations, (3)
focus groups (one child/youth participant group and peer-­reviewed articles in scientific journals, (4) organisa-
one parent/caregiver group at each site). Participants tion and institution newsletters and media releases and
18 years or older who provide independent consent (5) in accordance with the Australian National State-
to participate in the intervention will be asked if they ment 3.1.65, directly to participants and consumers in
would like the option of a parent/caregiver or support a format that is appropriate and accessible to them as
person to attend the parent/caregiver group. The the research will be likely to generate findings or results
aims of the qualitative interviews are to understand of significance to young people with CP and their fami-
how participants and/or their caregivers perceive their lies. The study will be reported in a way consistent with
involvement in the programme and elucidate barriers both Template for Intervention Description and Repli-
and facilitators to ongoing, sustainable participation cation57 and Consensus on Exercise Reporting Template
in Frame Running. Qualitative interview transciption guidelines.58
will be completed by a high-­quality paid service and
checked against original recordings. Participants will Author affiliations
have the opportunity to review their transcripts and 1
Child Health Research Centre, Faculty of Medicine, The University of Queensland,
edit their responses prior to analysis. Transcripts will Brisbane, Queensland, Australia
2
be thematically analysed using an inductive content Physiotherapy Department, Cairns and Hinterland Hospital and Health Service,
Cairns, Queensland, Australia
analysis approach.56 3
Institute for Musculoskeletal Health, School of Public Health, University of Sydney,
Sydney, New South Wales, Australia
Patient and public involvement 4
School of Human Movement and Nutrition Sciences, Faculty of Health and
A person with CP, a parent/caregiver and Frame Running Behavioural Sciences, The University of Queensland, Brisbane, Queensland,
organisations have been invited to participate as consumer Australia
5
Physiotherapy Department, Children's Hospital at Westmead, Sydney, New South
representatives during the study period. They will be Wales, Australia
financially compensated for their time and expertise at 6
Queensland Paediatric Rehabilitation Service, Department of Rehabilitation,
the rate of AU$50 per hour. A parent of a child with CP Queensland Children's Hospital, Brisbane, Queensland, Australia
(who participates and competes in Frame Running at an
international level) reviewed the protocol and provided Twitter Sarah E Reedman @sarah_reedman, Catherine Sherrington @cathiesherr,
feedback on the study design, which has been integrated. Emma Beckman @beckmanemma and Zoë Cotton @PhysiZo_Cotton
At least one consumer representative will meet with the Contributors SER, LS, LM, CS, EB, KW and RNB conceived the trial. SER completed
study team not less than every 2 months once recruitment the initial draft of the manuscript. MDC generated the randomization strata and
provided biostatistical advice and information. SGT, RT, and ID contributed technical
commences to provide advice and input in relation to expertise to the protocol manuscript for physical activity measurement, therapist
all following phases of the trial (conduct, analysis and outcome assessment, and Frame Running coaching respectively. AG and BD
reporting). designed the Frame Running training session content. ZC designed the assessment
methods and procedures. All authors designed the study, have read, edited, and
approved the final manuscript and supplementary files.
Funding The Run4HealthCP trial is financially supported by an Early Career
ETHICS AND DISSEMINATION Research Project Support Grant awarded by the Children’s Hospital Foundation
(grant ID: ECR0262020), the Merchant Charitable Foundation via the Children’s
Informed consent process Hospital Foundation (donation ID: 10415), and the Dr June Canavan Foundation
For children and youth <18 years of age or ≥18 years via the Children’s Hospital Foundation. Running frames used in the trial will
with an impaired capacity to consent, written informed be borrowed at no cost (except return freight) from various individuals and
consent will be obtained from the legal guardian. Youth organisations including the sole supplier.
≥18 years who can provide their own written informed Competing interests None declared.
consent will do so. This will occur after the treating/ Patient and public involvement Patients and/or the public were involved in the
assessing staff member has explained the study again in design, or conduct, or reporting, or dissemination plans of this research. Refer to
an accessible format (verbal, written) to the satisfaction the Methods section for further details.
of both the participating parent/guardian and/or child/ Patient consent for publication Not applicable.
youth. Provenance and peer review Not commissioned; externally peer reviewed.

10 Reedman SE, et al. BMJ Open 2022;12:e057668. doi:10.1136/bmjopen-2021-057668

 Open access

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