Background
Cerebral palsy (CP), the most common physical disability in childhood, is a group of disorders of the development of movement and posture that occur as a result of disturbances in the foetal or infant brain [
1]. The motor impairment may be accompanied by co-morbidities, including epilepsy, vision or hearing loss, intellectual disability, disorders of communication, behavioural difficulties, and secondary musculoskeletal problems [
1]. The most common motor disorder in CP is spasticity, occurring in 86 % of individuals [
2]. Spasticity is a velocity-dependent increase in the tonic stretch reflex, with exaggerated tendon reflexes [
3] and is characterised by slow, effortful movement [
4].
This research is embedded within an International Classification of Functioning, Disability and Health (ICF) framework [
5] that articulates a dynamic interaction between impairments at body structure and function level, activity performance and participation. At the body function level, muscle over-activity as result of spasticity and/or dystonia plays a significant role in the development of secondary musculoskeletal impairments in the upper limbs [
6] that are common in CP. Secondary impairments include muscle stiffness, loss of active range of movement, joint contracture and pain. Diminished skeletal muscle growth is a key feature in the aetiology of contracture and deformity [
6]. Persistent over-activity of skeletal muscle, and subsequent maintenance of a shortened position, can cause a failure of longitudinal muscle growth and muscle adaptation, including increased resistance to passive stretch or
stiffness [
6,
7]. Subsequently there is a biomechanical imbalance of bone to muscle, as bone continues to grow and muscle growth is impeded [
7]. The combined impact of these factors can result in soft tissue retraction, loss of active and passive range of motion and joint contracture [
6,
8].
Progressive changes in muscle length and stiffening of joints in the upper limbs can ultimately result in a limited ability to reach, grasp and manipulate objects or, in some individuals, a complete lack of functional use of the hands. Strong correlations exist between the degree of upper limb deformity and activity performance [
9]. When combined with neurological dysfunction, upper-limb musculoskeletal impairments significantly impact on the ability of children to use their hands to perform daily activities, attain age appropriate independence and develop the autonomy and skills required to participate in activities of importance in home, school and community environments [
10,
11].
Children with CP are not born with musculoskeletal impairments. There is evidence however, that in children with spastic motor types, these impairments begin to manifest prior to three years of age [
12] and that increasing stiffness and progressive loss of range of movement occurs throughout childhood and adolescence [
13‐
15].
A range of treatment options are available for children with CP that specifically focus on improving hand use. These include activity-based interventions such as goal-directed training, intensive bimanual therapy, modified constraint-induced movement therapy and home programs. Each of these interventions aim to achieve child/parent-focused goals, and has high-level evidence supporting their effectiveness for increasing activity-level performance and goal achievement [
16]. Little evidence is available however, about whether activity-level interventions improve range of movement and reduce secondary musculoskeletal impairments. In addition to activity-based therapies, injection of Botulinum toxin A (BoNT-A) into overactive muscle groups is known to reduce muscle overactivity and has been associated with improved range of movement during a period of chemical denervation, therefore enhancing the effects of upper extremity therapy and the potential for goal achievement and activity performance [
17]. Nevertheless, BoNT-A alone in the upper limbs has been shown to have little sustained effect on range of movement [
17]. Upper limb surgery is also available to correct deformity once present, although outcomes are variable [
18].
Removable orthoses (also called splints) are applied to the forearms, wrists and hands with the goal of either maintaining muscle length and joint range of movement through sustained stretch, or enhancing functional performance. Although orthoses are commonly integrated into intervention strategies with children with CP, there is little evidence supporting the use of upper limb orthoses and wide variation in their prescription, manufacture and intended aims [
10,
19]. One controlled trial [
20] demonstrated improved effect of BoNT-A when combined with static splinting (term used in the trial by Kanellopoulos et al.) in children with CP. A Cochrane systematic review by Katalinic et al. [
21], which included adults and children with a broad range of neurological and non-neurological conditions, demonstrated little benefit of stretching for preventing or reducing contractures. The review concluded that the use of interventions that provided a stretch to muscles, such as upper limb orthoses, be ceased [
21]. The application of these findings to children with CP however, is limited. Only five of the 35 randomised trials included children. Of these, three studies included children with CP and each of these evaluated the effects of casting (one in the upper and two in the lower limb) as opposed to orthosis wear. A more recent systematic review of the effectiveness of upper limb orthoses for children with CP found more equivocal evidence than Katalinic et al. and recommended further methodologically strong research be completed to more effectively inform practice [
22].
The clinical rationale for providing upper extremity orthoses is multi-faceted, with both short and long term goals. The focus of the current study is on rigid wrist/hand orthoses (WHO). The primary goal of wearing rigid WHO for all children with CP is to prevent the development of muscle stiffness, maintain the integrity of soft tissues and prevent the development of abnormal postures and long-term deformity. Due to the diverse nature of CP, the secondary goals generally depend on the child’s Manual Ability Classification System (MACS) level. The MACS is a five-level system that describes how children use their hands to handle objects during daily activities [
23]. For children in MACS levels I to III, who are able to handle objects in daily life, the secondary aims of WHO prescription are to improve or maintain activity performance and participation through maintenance of good posture for functional use of the hand. Children in MACS levels IV and V have little or no functional use of the upper limb(s) and the aim of wearing of a rigid WHO is to maintain posture to facilitate ease of care-giving during daily activities such as bathing, dressing and positioning. Wearing of a rigid WHO is also used to prevent complications associated with muscle shortening such as pain and poor palmar skin hygiene.
Application of the results of previous research has been limited by the length of time in which WHO have been applied and evaluated (often <6 months). These time frames are often dictated by the cost of implementing a trial. However, orthosis wear is an intervention aiming for long-term benefits (i.e. the reduction of contracture over time), thus clinicians are appropriately reluctant to change practice based on short-term research evidence. A well-designed large trial with a long intervention and follow-up period is now critical to determine the long-term outcome of this intervention. This 3 year trial has been designed to balance the need for longitudinal evidence with the complexities of attaining prolonged adherence of participants to an intervention within a controlled trial.
Effective and feasible rigid WHO are those where the benefits outweigh the risks associated with the intervention such as client discomfort, potential for skin breakdown, carer burden in maintaining routine application, follow-up appointments for manufacture and adjustment. Provision of WHO should also contribute positively to the long-term goals of children and families in terms of achievement of participation in meaningful activities during childhood and/or improved ease of caring for children with CP. Cost-effective rigid WHO are those where the benefits outweigh the net costs (defined as cost of the intervention minus the cost offsets) and/or where the relationship between the net costs and outcomes is deemed acceptable (i.e. less than a common decision threshold in Australia, such as <$50,000 per Quality Adjusted Life Year (QALY)). Affordable rigid WHO are those where the financial costs for materials, construction and monitoring are within the available budget of third party funders and/or parents.
Wrist-hand orthoses interventions, in combination with activity-based therapy, are aimed at maintaining muscle length, strength and balance, which are required for optimum force generation, effective grasp and manipulation [
24] and therefore functional use of affected hands in daily activities. The primary aim of this research is, therefore, to evaluate whether use of rigid WHO over 3 years in children with CP, combined with usual multidisciplinary care, can prevent or reduce musculoskeletal impairment including loss of range of movement and muscle stiffness at the wrist, compared to usual multidisciplinary care alone. The impact of WHO wear on pain, activity performance and participation, as well as ease of caregiving for families will be evaluated along with an assessment of cost-effectiveness of the intervention.
Data collection and analyses
Assessors and details of blinding
Assessors will be occupational therapists or physiotherapists blinded to the treatment group of the child. Each assessor will be trained by a chief investigator in reliable administration of all measures and provided with a study assessment protocol to ensure consistency of assessment techniques between assessors. Multiple efforts will be made to retain blinded status of assessors. Treating therapists, research personnel, child-participants and families will be routinely reminded that it is critical that blinding of the assessor is retained and the assessor will remind the family of this on initial contact at each assessment. Study numbers allocated to children will not contain a fixed code denoting intervention group. The success of blinding will not be measured as methodological experts argue against such practice [
28]. Therapists providing the WHO are unable to be blinded to treatment group and hence may adjust co-interventions differently in the two groups. Measurement of co-interventions and comparisons between groups is therefore is an important part of this trial. Blinding of parents and participants to intervention group is not possible.
Demographic and diagnostic characteristics
Consistent with best practice in CP [
1], the severity of CP will be assessed and classified at baseline using the Gross Motor Function Classification System [
29], Manual Ability Classification System [
23], Communication Function Classification System [
30] and Bimanual Fine Motor Function scale [
31]. In addition, the type of movement disorder (that is, spastic or mixed) and severity of spasticity in the included upper limb(s) will be rated using the Hypertonia Assessment Tool [
32] and the Australian Spasticity Assessment Scale [
33,
34] respectively. Information from these tools will help to describe the characteristics of the study sample and be used in post-hoc analyses of outcomes. A study specific questionnaire will measure a range of demographic and other variables including age, gender, associated conditions (intellectual disability, sensory impairments), family configuration, range of services received and socio-economic status as defined using the Socio-Economic Index for Areas data [
35].
Outcome measures
The primary outcome measure is passive range of wrist extension (measured with the fingers extended). Range of wrist movement is operationalised from -70° (full wrist flexion) to + 80° (full wrist extension) where 0° indicates a neutral position. Range of movement will be measured using a goniometer for extension/flexion movements, an inclinometer for supination/pronation and inertial motion sensors. Inertial motion sensors, constructed specifically for the trial, will be used to measure active and passive wrist extension, with fingers extended as well as functional range of wrist extension elicited during standardised tasks. Other measures across the domains of the ICF will also be used to evaluate the effect of the intervention. Table
2 displays each variable, the measurement tool selected and provides an overview of psychometric evidence for the selected tools.
Table 2
Variables and outcome measures
ICF level: Body function: baseline, 6, 12, 18, 24, 30, 36 months |
Passive range of motion: elbow extension, wrist extension (with fingers extended), wrist extension (with fingers flexed), supination | Standardised goniometric measurement; inclinometer for measures of supination; | Goniometric measurements have a high level of intra-rater reliability when measuring passive range of movement in the lower limb in children with CP (ICC >.80) and SEM of 3.5° [ 44, 45]. |
Inertial Motion sensors. | Inertial motion sensors (see additional information below) will be used to measure passive wrist extension with fingers extended only. |
Active range of movement: elbow extension, wrist extension (with fingers extended), wrist extension (with fingers flexed), supination | Standardised goniometric measurement and use of inclinometer for measures of supination. | See additional information above. |
Inertial Motion sensors | Inertial motion sensors (see additional information below) will be used to measure active wrist extension with fingers extended only. |
Functional range of wrist extension during standardised tasks. | Inertial Motion sensors. | A wireless inertial motion sensor for children has been designed and engineered for this trial to measure wrist flexion/extension movement during functional activity. The sensors use a combination of inertial sensor technologies to provide an accurate estimate of orientation referenced to a fixed frame [ 46]. Once correctly positioned they wirelessly capture movement with 3° of freedom in a virtual reality environment to provide continuous kinematic data during unrestricted functional movements. The validity and reliability of the newly developed sensor has been assessed with 10 children with CP (aged 4–12 years) against 3DMA, the ‘gold standard’ method to quantify movement. Preliminary data demonstrates the inertial motion sensors have excellent static and dynamic accuracy (+/-0.5 and +/-1.2° respectively). |
Muscle stiffness (finger flexors, wrist flexors, pronators and elbow flexors) | Modified Ashworth Scale [ 47] | The six point Modified Ashworth Scale has moderate intra-rater reliability when assessing the elbow (ICC 0.66) and wrist flexors (ICC 0.57) in children with CP [ 48]. |
Muscle spasticity (finger flexors, wrist flexors, pronators and elbow flexors) | Modified Tardieu Scale [ 47] | The Modified Tardieu Scale has moderate to high intra-rater reliability when assessing the elbow (ICC 0.65) and wrist flexors (ICC 0.92) in children with CP [ 48]. |
Australian Spasticity Scale [ 33] | The Australian Spasticity Assessment Scale has demonstrated moderate to high inter-rater agreement (47–100 %) [ 33] |
Grip strength | Hand held dynamometer (CITEC) | Dynamometery has been shown to have excellent levels of inter-rater (ICC 0.95) and test-re-test reliability (ICC 0.96) when measuring strength in the hand of children with hemiplegic CP [ 49]. |
Hand deformity | Neurological Hand Deformity Classification Scale [ 50] | The Neurological Hand Deformity Classification has evidence of reliability for children with spastic cerebral palsy with high inter-rater agreement (Kappa 0.87) and intra-rater agreement (Kappa 0.91) [ 15] |
Thumb position | House Thumb in Palm classification [ 51] | This measure has been developed for children with CP based on the predictors of surgical success and has been found to be reliable: Kappa = 0.73 (rater agreement) and 0.74 (test-re-test agreement) [ 49, 52]. |
Hand pain | Study specific questionnaire | The study specific questionnaire was developed for this study to document parent perception of domains unable to be captured in existing measures. Questions will be completed by the child where possible or by a parent/carer proxy. Although proxy respondents are known to underestimate pain, parent-reported pain will be required for children who are more severely cognitively impaired or unable to communicate their pain effectively. |
Activity domain of the ICF: baseline, 12, 24 & 36 months |
Self-care skills | Pediatric Evaluation of Disability Inventory – Computer Adaptive Test [ 53] | This is a standardised assessment of how children with impairments function in the context of their daily life. The Pediatric Evaluation of Disability- Computer Aided Test provides an accurate and precise assessment of abilities in four functional domains (ICC 0.99). For this trial only data from the Daily Activities domain will be collected. |
Manual ability | | This tool has been Rasch analysed and has demonstrated validity and appropriate range and measurement precision for clinical practice and research: reliability: R = 0.94; reproducibility over time: R = 0.91 [ 54]. |
Speed and dexterity | | This test has a high level of intra-rater (ICC 0.99) and test-retest reliability (ICC 0.85) [ 56]. |
Hand function | Modified House Scale [ 57] | This scale is reliable in children with CP: inter rater reliability (ICC 0.94-0.96); intra rater reliability (ICC 0.93-0.96) [ 57]. Rasch analysis was performed on the original scale and the items reduced: analysis suggests that the modified version demonstrates good construct validity [ 58]. |
Ease of care-giving | Study specific questionnaire | Parent response to specific questions regarding the child’s ability to use their hands in self-care tasks or, for children with severe forms of cerebral palsy the ease with which parents or carer’s can complete daily tasks of care for them. |
Participation domain of ICF: Baseline & 3 years only |
Participation | Participation and Environment Measure-Child & Youth [ 59] | Designed to measure frequency of participation, involvement during participation and the impact of the environment on participation in children aged 5 to17 years [ 59]. This measure captures participation outcomes in home, school and community contexts. Reliability of the frequency scales (ICC range 0.58-0.84) and involvement scales (ICC 0.69-0.76 is moderate to high [ 59]. |
Child Health related quality of life and care-giving burden | Cerebral Palsy Quality of Life Questionnaire – Child and Teen versions [ 60, 61] | Due to the varying ages and abilities of the child-participants, both parent- and self-report versions of the Child or Teen CP Quality of Life will be used to measure quality of life. Test-re-test reliability for the Child version was high (ranged from ICC 0.76 to 0.89 across 7 scales) [ 61], and moderate to high for the Teen version (ICC 0.57 to 0.88) [ 60] |
Health economic measures: Baseline, 12, 24, 36 months |
Cost Effectiveness Analysis (CEA) | Study specific questionnaire | Data on type and number of health professional appointments attended by child in preceding 6-month time period will be utilised for calculation of healthcare cost as well as out of pocket costs to families. Net incremental costs expressed as ICER to meaningful clinical and physical outcomes (e.g. selected from body function domains; activity domains; and the clinical quality of life questionnaire). |
Cost Utility Analysis (CUA) | Child Health Utility -9 Dimensions [ 37] | Net ICER to the quality of life improvement for children and parents/carers expressed as QALY using an economic MAUI. Where possible the Child Health Utility will be completed along with the parent proxy version. The Child Health Utility has 9 items, takes 2-3 min to complete and covers worry, sadness, pain, tiredness, annoyance, school work, sleep, daily routine and ability to join in activities. The Child Health Utility-9D demonstrated good validity and high levels of agreement with a similar instrument (ICC: 0.742) [ 62]. The parent measure of quality of life, the Assessment of Quality of Life 8 Dimensions has high reliability (ICC 0.89) [ 36]. |
Assessment of Quality of Life 8 Dimensions [ 36] |
Cost Consequences Analysis (CCA) | | CEA/CUA reported alongside a broader documentation of child & family relevant outcomes |
Economic analysis
Economic analysis in the context of trials is designed to answer one or both of two questions: i) does the treatment being evaluated offer value-for-money (i.e. ‘allocative efficiency’); and ii) if so, how best to design/implement it (i.e. ‘technical efficiency’). In this trial we are focussed on technical efficiency. Specifically, is the care pathway more cost-effective with the addition of a WHO for children with CP than without? Economic methods have been chosen therefore to focus on appraisal using trial-based data (with limited economic modelling) and to deal with variability in usual multidisciplinary care. A comprehensive analysis of usual care activities will enable: i) specification of a weighted average usual care pathway (i.e. weighted by activity prevalence); ii) incremental cost-effectiveness ratios (ICERs) presented by state/site, as well as by overall trial results; and iii) extensive sensitivity/uncertainty analyses to detail cost and outcome drivers.
The technical efficiency focus will make cost-effectiveness analysis (CEA) and cost consequences analysis (CCA) the primary analysis, with ICERs that focus on body function, activity measures and child/family quality of life and health utility outcomes from both health sector and service funder perspectives. The key trial-based ICERs are: i) the ‘net cost per 10° improvement in passive range of movement at 3 years’; ii) the ‘net cost per unit of improvement on the
Cerebral Palsy-Quality of Life’ measure; iii) the ‘net cost per
Adult Quality of Life-8 Dimensions’ [
36] improvement for parent/carer; and iv) the ‘net cost per
Child Health Utility 9D’ [
37] improvements for children. While economic quality of life instruments are usually focussed on value-for-money comparisons, the difficulties in modelling longer term outcomes in this trial, leads to their primary purpose being to help establish technical efficiency. ICERs will be reported as both point and range estimates. In the CCA, ICERs will be reported and interpreted alongside the full range of body function and activity measures collected. A Cost-Utility Analysis (CUA) with variable time horizons and best available data will be included in sensitivity analysis against a specified decision threshold (i.e. < $50,000 per QALY). In addition to the CEA/CCA and CUA, a broader economic approach will be undertaken to capture policy and implementation/policy issues (e.g. acceptability to stakeholders, equity impacts, feasibility of implementation, quality of the evidence base) using
Assessment of
Cost
Effectiveness (
ACE) methods.
ACE has been used across a series of commissioned and NHMRC-funded projects [
38].
Costs will be calculated using pathway analysis to document treatment activity, specify unit prices and estimate costs and potential cost offsets across the study groups. For usual multidisciplinary care, a number of pathways will be constructed and analysed separately as well as a weighted average comparator. Costs associated with the WHO will be assessed by expenditure category (i.e. salaries, overheads, consumables) with economic data collected using a logbook; all other healthcare costs will be assessed by incidence category (i.e. who bears the cost) using available information from sources such as the Medical Benefit Schedule and Pharmaceutical Benefit Scheme. Sensitivity/uncertainty analyses will be undertaken to investigate the robustness of the ICERs to variations in key cost, pathway and outcome parameters in the trial and across sites.
Data collection methods for child-participants who exit prematurely
Children may exit prematurely from the study because of voluntary withdrawal or termination of WHO intervention due to harm (e.g. allergic reaction to materials). Participant retention will be supported within the study through provision of routine follow-up and regular feedback on child progress via the TherApp summary report that can be generated by parents throughout the trial. Where possible, all children will be followed to the study end point (3 years) so that data are available for analyses. Lack of adherence to the treatment plan will be recorded using TherApp and will not constitute a reason for withdrawal. Reasons for withdrawal from the intervention, or the study, will be recorded to assist with management of missing data and interpretation of results.
Monitoring of harm and adverse events
No harm or adverse events from orthoses are reported in the literature but are occasionally noted in clinical practice; these are temporary and non-sentinel. Harm arising from the WHO could include the development of pressure areas on the skin, pain, disturbed sleep or behaviour, and skin allergies from specific splint materials while wearing the orthosis, and heat during orthosis fabrication. Children in both groups are at risk of a reduction in joint range of movement as part of the natural course of CP during growth and development. Adverse events unrelated to the study may also occur and will be adjudicated by the Data Monitoring Committee. Data related to harm and/or adverse events for all children will be collected throughout the study by the therapist who manufactures the WHO (routine follow up), retrospectively by study research assistants (6 monthly) and via TherApp alerts. If the TherApp registers an adverse event an automated email alert to the study research assistant will enable appropriate follow up.
Data management
Data will be collected using a combination of paper-based and web-based data forms supported by the secure Research Electronic Data capture (REDCap) data management system [
39] hosted at the Murdoch Childrens Research Institute. REDCap supports quality control measures including rule-based data entry to reduce data entry errors. In addition, data cleaning will be undertaken. Secure electronic data storage will be undertaken using REDCap, and secure (locked) local storage of original paper-based versions of data collected will occur in accordance with ethically approved procedures for each trial site. Participants will be assigned identification codes on enrolment to the study. These codes will be used during data entry so that data are de-identified during analyses and only aggregated data reported to protect the privacy of participants.
Statistical methods
The primary analysis will be by intention to treat. Comparison between the intervention and the control groups in the difference from baseline in the passive range of wrist extension (primary outcome) will be presented as the mean difference between the groups and its 95 % confidence interval, obtained using linear regression adjusted for the stratification factors of site and range of passive wrist extension at baseline. The regression model will be fitted using generalised estimating equations (GEE) to allow for the clustering of observations within children for those with both limbs in the study. To explore the effect of the adherence to WHO wearing schedule (i.e. a dose response relationship), a linear regression model will be fitted with compliance to treatment as a predictor and difference in the passive range of wrist extension from baseline to 36 months as the outcome, applied to all study participants. Again this model will be fitted using GEEs to allow for the clustering of limbs within participants. Evidence for an interaction between age and treatment, and between severity (Neurological Hand Deformity Classification) and treatment, will be explored by the inclusion of interaction terms in the linear regression models as well as GEE models. The analyses will be repeated, adjusting for potential confounders including occasions of upper limb BoNT-A injections and frequency of upper limb intervention. Analysis will also be undertaken using the ‘per protocol’ population excluding children who received surgical intervention or casting during the study. All data available from children who are withdrawn from the study prior to study completion will be used for analysis. Imputation of missing data will only be considered in the primary analysis if less than 10–20 % of the primary outcome is missing and will be undertaken throughout multiple imputation models.
Depending on whether data are continuous, categorical or dichotomous, the appropriate generalized linear model will be used to estimate the effect of treatment across the study period on secondary outcomes, again fitted using generalised estimating equations. All analyses will be adjusted for the same stratification factors as for the primary analysis and carried out on intention to treat and per protocol populations.
Discussion/conclusion
Hand dysfunction and deformity are prevalent in CP: 85 % of children have spasticity that impacts upper limb structure and function, ≥62 % have wrist flexion deformities, and early onset is common [
41]. Strong positive correlations exist between hand posture and function [
9,
41]. Hand orthoses are time-consuming to make and are challenging for families to implement and for children to wear, but if they prevent deformity and improve hand function, they are a vital treatment. This RCT should provide high quality evidence to resolve the long debate about the value of WHO and the specific impact of wrist impairment on activity. In addition, three novel measurement devices will be designed and/or engineered, tested and validated in children within the conduct of this trial: (i) TherApp; (ii) within-orthosis tactile sensors; and (iii) inertial motion sensors. The further application of these devices in a diverse range of clinical and research contexts will constitute a significant intellectual and practical contribution to the health sciences. TherApp has potential for application to support data collection in other health intervention research trials and in clinical practice to support the implementation of interventions and facilitate communication between clients and clinicians. Inertial motion sensors have potential applicability to other interventions focused on outcomes at the body structure and function level of the ICF, such as BoNT-A, and the tactile sensors may also provide data about orthosis fit as well as wearing time, if placed within the orthosis at key points of hand-orthosis contact.
The annual cost of CP in Australia is approximately $1.5 billion (0.14 % of GDP) [
42]. Lost wellbeing (as a result of disability and premature death) can be valued at a further $2.4 billion [
42]. This research will provide Level II (RCT) evidence [
43] to inform clinicians, health services, government funding bodies and parents and carers of children with CP whether the provision of orthoses to prevent upper limb impairment is worth the effort and associated costs. This multicentre RCT along with a companion RCT to be implemented with young children under the age of 3 years will provide high quality evidence of the medium-term effect of rigid upper limb orthoses in children with CP. The second trial aims to determine whether provision of a rigid WHO can prevent the occurrence of contracture and deformity in children aged less than 3 years at time of recruitment. By combining the use of rigid orthoses with usual multidisciplinary therapies, these two trials will investigate a combined intervention more reflective of current best practice than has been previously investigated. The results will provide evidence as to whether the use of rigid upper limb orthoses are needed, or if activity-based therapy alone is sufficient to restore and prevent musculoskeletal impairment in children and adolescents with CP.
Acknowledgements
Ms Jacqui Wisemantel and Ms Melissa Weston for their support and expertise as parent advisers to the trial.
Dr Katherine Lee, Centre for Epidemiology and Biostatistics at the Murdoch Childrens Research Institute for support with statistical elements of the trial.
Mr Simon Garbellini for his contribution to the development of the consensus based guidelines for WHO prescription.
Dr Iain Murray, Mr Weiyang Xu, Dr Cesar Ortega-Sanchez, Department of Electrical and Computer Engineering, Curtin University and Dr Sian Williams, Dr Tifffany Grisbrook and Corrin Walmsley, School of Physiotherapy and Exercise Science, Curtin University for the development of the inertial motion sensors.
Competing interests
The authors declare they have no competing interests.
Authors’ contributions
CI, MW, CE, BH, SG, MR and BA conceived the study and initiated the study design and implementation. All authors collaborated on the development and refinement of the study protocol and have read and approved the final version. MR is the National Project Coordinator and is based in Victoria. MW will take responsibility for the study implementation in New South Wales. CE will take responsibility for study implementation in Western Australia. BH and SG will take responsibility for study implementation in Victoria. DR is the lead investigator on the CRE-CP which nominated this study as a key area for investigation. RC and SS are responsible for the design, implementation, analysis and interpretation of the health economics component of the study. FO will lead the statistical analysis and data management components. All authors have read and approved the manuscript.