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Performance of school-aged children with cerebral palsy at GMFCS levels I and II on high-level, sports-focussed gross motor assessments

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Performance of school-aged children with cerebral palsy at GMFCS levels I and II on high-level, sports-focussed gross motor assessments

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Purpose: To investigate performance of children with cerebral palsy (CP) at GMFCS Levels I-II on sports-focussed gross motor assessments; and concurrent validity of mainstream and CP-specific high-level assessment batteries and field tests.

Methods: Fifty-four children (6-12 years) with CP at GMFCS I-II completed the Test of Gross Motor Development-second edition, Gross Motor Function Measure-Challenge Module, Muscle Power Sprint Test (MPST), 10 × 5 m Sprint Test (10 × 5 mST), Vertical Jump, Broad Jump, and Seated Throw. Correlations between measures, age and mobility level, and group differences between age and mobility level were examined and content analysis performed.

Results: Children at GMFCS I demonstrated significantly higher gross motor assessment battery scores than children at GMFCS II ( U = 73.5-109.0, p < 0.001). Performance improved with age for children at Level I but not II. Children with higher overall motor scores scored higher on running (MPST, 10 × 5 mST, r = -0.516 to -0.816, p < 0.001), jumping (Vertical Jump, Broad Jump, r = 0.499-0.774, p < 0.001) and throwing (Seated Throw, r = 0.341-0.500, p  = 0.012 < 0.001) field tests.

Conclusions: High-level gross motor assessments were achievable and appropriately challenging for children with CP at GMFCS I-II. Scores discriminated between performance and were associated with mobility level. Concurrent validity was established between gross motor assessment batteries, and locomotor field tests.

IMPLICATIONS FOR REHABILITATION

For children with cerebral palsy at GMFCS I-II, sports-focussed assessments should be used to assess high-level gross motor function.

The Gross Motor Function Measure- Challenge and Test of Gross Motor Development demonstrate no ceiling for children with cerebral palsy at GMFCS I-II.

Single-item running and jumping field tests provide targeted skill assessment and estimate sports skills for children with cerebral palsy.

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Performance of school-aged children with cerebral palsy at GMFCS levels I and II on high-level, sports-focussed gross motor assessments

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Purpose: To investigate performance of children with cerebral palsy (CP) at GMFCS Levels I-II on sports-focussed gross motor assessments; and concurrent validity of mainstream and CP-specific high-level assessment batteries and field tests.

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Radio interview regarding research into breaking down barriers to participation in physical activity for children with disabilities. Media related to public lecture provided as part of CSU's exploration series.

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SPORTS Participation Framework for children with disability

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SPORTS Participation Framework for children with disability

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Published in the Sports Stars protocol paper, BMC Paediatrics 2018

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Poster presentation at 2017 American Academy of Cerebral Palsy and Developmental Medicine Conference in Montreal

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Poster presentation at 2017 American Academy of Cerebral Palsy and Developmental Medicine Conference in Montreal

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Active exercise interventions improve gross motor function of ambulant children with cerebral palsy: A systematic review

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Active exercise interventions improve gross motor function of ambulant children with cerebral palsy: A systematic review

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Published in Disability and Rehabilitaiton

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PURPOSE:

Evaluate effectiveness of active exercise interventions for improving gross motor activity/participation of school-aged, ambulant/semi-ambulant children with cerebral palsy (CP).

METHOD:

A systematic review was conducted following PRISMA guidelines. Five databases were searched for papers including school-aged children with CP, participating in active, exercise interventions with gross motor outcomes measured at the Activity/Participation level. Interventions with previous systematic reviews were excluded (e.g. hippotherapy). Evidence Level and conduct were examined by two raters.

RESULTS:

Seven interventions (34 studies) met criteria. All studies reported on gross motor function, however, a limited number investigated participation outcomes. Strong positive evidence was available for Gross Motor Activity Training (n= 6, Evidence Level II-IV), and Gross Motor Activity Training with progressive resistance exercise plus additional physiotherapy (n = 3, all Evidence Level II). Moderate positive evidence exists for Gross Motor Activity Training plus additional physiotherapy (n = 2, all Evidence Level II) and Physical Fitness Training (n = 4, Evidence Level II-V). Weak positive evidence was available for Modified Sport (n = 3, Evidence Level IV-V) and Non-Immersive Virtual Reality (n = 12, Evidence Level II-V). There was strong evidence against Gross Motor Activity Training plus progressive resistance exercise without additional physiotherapy (n = 4, all Evidence Level II).

INTERPRETATION:

Active, performance-focused exercise with variable practice opportunities improves gross motor function in ambulant/semi-ambulant children with CP. Implications for rehabilitation Active exercise interventions improve gross motor function of ambulant/semi-ambulant children with cerebral palsy. Gross Motor Activity Training is the most common and effective intervention. Practice variability is essential to improve gross motor function. Participation was rarely measured and requires further research, particularly in interventions that embed real-world participation opportunities like Modified Sport.

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Active exercise interventions improve gross motor function of ambulant/semi-ambulant children with cerebral palsy: a systematic review.

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PURPOSE: Evaluate effectiveness of active exercise interventions for improving gross motor activity/participation of school-aged, ambulant/semi-ambulant children with cerebral palsy (CP). METHOD: A systematic review was conducted following PRISMA guidelines. Five databases were searched for papers including school-aged children with CP, participating in active, exercise interventions with gross motor outcomes measured at the Activity/Participation level.

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SPORTS STARS study protocol: a randomised, controlled trial of the effectiveness of a physiotherapist-led modified sport intervention for ambulant school-aged children with cerebral palsy" 

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SPORTS STARS study protocol: a randomised, controlled trial of the effectiveness of a physiotherapist-led modified sport intervention for ambulant school-aged children with cerebral palsy" 

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Open access in BMC Paediatrics

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Study aims

The proposed study is a randomised, waitlist controlled, assessor blinded, superiority trial with two parallel groups. This study will aim to compare the effectiveness of a group-based, sports oriented physiotherapy intervention, Sports Stars, for ambulant school-aged children with CP, to standard care across all ICF domains. This study will also aim to gain feedback from treating physiotherapists and caregivers involved in the study to determine the acceptability of the intervention. The specific hypotheses to be tested are:

Primary hypothesis

H1: Compared to children in the standard care group, children in the Sports Stars group will demonstrate greater improvement in their individually-selected, sports activity and participation related goals measured by the Canadian Occupational Performance Measure (COPM).

Secondary hypotheses

H2: Body function and structure: Compared to children in the standard care group, children in the Sports Stars group will demonstrate greater improvements in lower limb and upper limb strength, balance, agility and their aerobic and anaerobic fitness.

H3: Activity: Compared to children in the standard care group, children in the Sports Stars group will demonstrate greater improvements in gross motor capacity, including locomotor ability and object control skills.

H4: Participation: Compared to children in the standard care group, children in the Sports Stars group will demonstrate increased participation in physical activities including recreational or formal sporting activities.

H5: Quality of life: Compared to children in the standard care group, children in the Sports Stars group will demonstrate greater improvements in quality of life as measured by parent report.

Study sample and recruitment

Inclusion criteria

This study will include children who:

1. 1.
Are aged 6-12 years at study entry;

2. 2.
Have a confirmed diagnosis of CP;

3. 3.
Are ambulant without aids (classified as GMFCS- E&R Level I or II);

4. 4.
Can commit to eight, one-hour weekly group physiotherapy sessions and three, two-hour assessment appointments over a period of 6 months.

Exclusion criteria

Children will be excluded from the study if they:

1. 1.
Are unable to complete baseline assessments;

2. 2.
Have had orthopaedic or neurological surgery within 6 months prior to the immediate intervention start date;

3. 3.
Have had Botulinum Toxin injections within 3 months prior to the immediate intervention start date;

4. 4.
Have intellectual or behavioural difficulties which would limit their ability to participate in the assessment or therapy protocols;

5. 5.
Have medical co-morbidities which prevent them from exercising safely (e.g. cardiac or respiratory instability, uncontrolled seizures).

Criteria for withdrawal

Participants will be excluded from the study if they fail to attend either their baseline assessment or withdraw prior to the commencement of the immediate Sports Stars group. Primary analysis will use the intention to treat principle, using the last observation carried forward for participants who withdraw after commencement of intervention in the immediate Sports Stars group.

Recruitment

Sample size

According to CONSORT guidelines, sample size calculations are based on adequate power for comparison between the effects of the Sports Stars program compared to standard care using the COPM immediately post intervention (T2). Data from a previous study investigating the effects of an exercise group aiming to improve physical activity, balance and strength in ambulant children with CP (6-14 years) showed a standard deviation of 1.87/2.88 (intervention/control) [ 32]. This standard deviation and a mean change of 2 points for performance on the COPM (clinically meaningful difference) were used to calculate sample size. Based on significance (alpha) of 0.05 and 80% power, a minimum sample of 25 participants in each group (50 participants total) will be required. Therefore, 60 participants (30 in each group) will be recruited to allow for 20% attrition.

Recruitment process

Eligible children will be prospectively recruited through the client database of a state-wide community rehabilitation service. As geographical location and participant availability are critical to forming groups of sufficient participant numbers for this study, participants will be recruited in blocks according to their geographical location. Once at least four, and a maximum of six, children are identified that can attend an eight-week group on a specified day in a specific geographical location, these children will be assigned as group one. When a second group of four to six children are identified for a geographical location, they will be assigned as group two. When two groups of four to six participants each are identified, they will proceed to randomisation.

Randomisation

A random sequence will be generated via coin flip by an independent, off-site co-investigator (MA) who will not be involved with assessment or treatment (as per process used in previous studies [ 17]). The outcome (heads: group 1 = immediate Sports Stars intervention, group 2 = waitlist Sports Stars intervention. Tails: group 1 = waitlist Sports Stars intervention, group 2 = immediate Sports Stars intervention) will be written on a piece of paper and concealed inside a sequentially numbered, opaque envelope and stored securely off-site.

When two groups are identified, they will be randomly assigned to either the immediate intervention group or the waitlist control group with a 1:1 block allocation via drawing of one of the opaque envelopes. Randomisation will be completed offsite by the same independent co-investigator (MA). This process will continue until 60 participants complete baseline assessments and proceed to the Sports Stars group.

Therapy protocols and delivery

Refer to Fig. 2 for the study flow diagram according to CONSORT guidelines.

Sports Stars flow chart according to CONSORT guidelines

Sports Stars intervention

Each group will contain between four and six participants with one lead Physiotherapist. The immediate Sports Stars group will receive eight, one-hour, weekly sessions (8 hours) of group-based, sports specific fundamental movement skills training, detailed in Fig. 3. These groups will introduce children to four popular Australian sports and support the development of core motor skills for transition to recreational mainstream and modified sports opportunities including Junior Entry Point sports programs, NetSetGo (netball), In2Cricket, MiniRoos (soccer) or T-Ball (softball/baseball). To achieve this, Sports Stars will focus on developing key Body Functions (aerobic and anaerobic fitness, muscle strength, balance and agility, and locomotor and object control) and sport-specific Activity skills to facilitate participation in modified sport games of soccer, netball, T-ball and cricket.

Sports Stars session content summary

Location

Sports Stars will be conducted at local parks in the community in association with Queensland's largest state-wide community physiotherapy service provider for children with CP. Groups will be provided in urban/regional Queensland, Australia, capturing the breadth of cultural and socioeconomic diversity. Each group will include back-up undercover areas to be used in the event of wet weather.

Participants of the Sports Stars group will not be permitted to receive other physiotherapy during their 8 week Sports Stars group. Throughout the duration of the study, all participants will be permitted to access standard care from other allied health (e.g. occupational therapy, speech and language pathology) and/or other adjunct therapies. Parents/guardians will be requested to record the frequency, duration and content of any standard care received including any home exercise performed.

Standard care comparison

Participants in the Sports Stars Waitlist group will receive standard care. This describes the typical intervention that a six to 12-year-old child with a diagnosis of CP would receive from their normal therapists in community, private or hospital contexts. Therapists who provided standard care will have varying degrees of experience working with children with CP. Standard care may include neurodevelopmental therapy, context-focussed therapy, strength or fitness training, or functional training. At this age, children are typically seen for 1:1 therapy, however some group therapy may also be offered. Ambulant children of this age (classified as GMFCS- E&R I-II) with CP would typically receive only 1-2 physiotherapy sessions per quarter. This may include a home program and is expected to vary between participants, however most children would receive no more than two sessions during the comparative 8 weeks of a Sports Stars program. Details of frequency, duration and content of any therapy session, particularly physiotherapy, will be collected via a parent log book throughout the project.

Treatment fidelity

Intervention therapists will be masked to baseline outcome assessments. All intervention therapists providing the Sports Stars intervention will be qualified physiotherapists with experience working with children with CP in a community setting. At therapist training, the detailed Sports Stars protocol and a sports equipment pack will be provided to all therapists to ensure that participants in all groups received consistent delivery. This includes a written week by week program of sports focussed exercises and progressions (Additional file 1) with detailed instructions and video resources that therapists can use to select predetermined difficulty levels to suit each child's age, goals and preferences. Therapists will receive individual, face to face training in the Sports Stars protocol by the first author prior to the commencement of the group. Treating therapists will engage in further consultation as required with the study coordinator either by phone or in person to review the Sports Stars package and to discuss program content and structure prior to providing the group to participants. Each child will have features of their participation in each group session recorded by the treating therapist, including exercise type, level of difficulty and therapist observed engagement. At week three and six of the study, each therapist will undergo fidelity review and receive guidance via email through an independent, offsite co-investigator (MA) who will not be involved with assessment or treatment regarding intervention format, content, progression and data recording. Independent content analysis will determine compliance with the provided protocol. Parents of both the immediate and waitlist groups will be provided with training journals to record any therapy, including home exercises, and sport that they undertake during the study period.

Outcome measures and procedures

All outcomes will be collected pre-intervention, immediately post intervention and at 12 weeks post intervention by the blinded chief investigator (GC).

Participants will be classified based on their severity and age to compare groups at baseline. They will be classified according to:

The Gross Motor Function Classification System is an internationally recognized classification system for children and youth with CP. Participants will be classified as either classified as GMFCS- E&R I (able to walk independently at home, school, outdoors and in the community with limitations in speed, balance or coordination) or II (able to walk in most settings, however may have difficulty with distances or more challenging environments and gross motor skills like running and jumping) using the descriptors for between the child's 6th and 12th birthday [ 33].

Participants will be classified by motor type (spasticity, dyskinesia, ataxia or unclassifiable) and distribution (unilateral or bilateral) [ 34, 35].

Participants will be classified by their ability to walk five, 50 and 500 m, correlating to their ability at home, school and community. Scores range from N (does not apply), to 6 (independent on all surfaces without aids) [ 36].

2.
Outcomes: Measured for all participants at four time points and compared to baseline and each other:

Time one (T1): zero months- Baseline

Time two (T2): 2 months- Immediately after immediate intervention

Time three (T3): 5 months- 12 weeks after immediate intervention

For children participating in the waitlist group, an optional fourth assessment will occur:

Body function and structure outcomes

Aerobic capacity & agility: 10 × 5 Meter Sprint Test [37]

The 10 × 5 Meter Sprint Test is designed to measure aerobic capacity and agility in children with CP of GMFCS- E&R level I or II. Children must continuously sprint the five-meter course 10 times, making turns at the cones marking the end of the five meters. The 10 × 5 Meter Sprint Test has excellent inter-observer (ICC > 0.97) and test-retest reliability ( r = 1). It has reported good construct validity. The 10 × 5-m sprint test is sensitive to change for children at GMFCS- E&R levels I and II and therapists report a high clinical feasibility. A decrease in exercise time of 3.2 s would be considered real change [ 37].

Anaerobic Capacity Muscle Power Sprint Test (MPST) [37]

The MPST measures anaerobic capacity by asking the participant to sprint 15 m (marked by lines and cones) at their maximum pace, 6 times, with 10 s recovery between each sprint. The MPST has a high inter-observer and test-retest reliability ( r = 0.97-0.99). It has good construct validity with GMFCS- E&R [ 37] and the Wingate Anaerobic Test (Peak Power: r = 0.731, Mean Power: r = 0.903) [ 38]. It is sensitive to change in children GMFCS- E&R level I and II and has high clinical feasibility. Standard errors of measurement were reported at 13.9 (peak power) and 9 (mean power) Watts [ 37].

Functional Lower Limb Strength: Standing Broad Jump [39]

The standing broad jump measures lower limb strength in the context of sports participation. Standing with toes up to a line, children are asked to jump forward as far as they can, landing with both feet. The distance between the start line and the most distal part of their toes of their back foot will be measured for three jumps, with the average recorded to the nearest centimetre. The standing broad jump has excellent test-retest reliability in typically developing children (ICC- 0.88 [ 40]) and in children with down syndrome (ICC- 0.89 [ 41]). It has excellent concurrent validity with measures of physical fitness ( r = 0.84) [ 40], Paralympic throwing ( r = 0.77-0.86) [ 42] and sprinting ( r = 0.82) [ 43] for children with disabilities. It has been reported to be sensitive to change as part of a test battery and is a feasible clinical test [ 40].

Functional Lower Limb Strength: Vertical Jump [39]

The vertical jump measures lower limb strength. Standing next to a wall, children raise their arm. The most distal point of their fingers is marked. They are instructed to jump as high as they can, a second mark being made at the height of their jump. The vertical distance of three jumps will be measured and averaged to obtain the jump height to the nearest centimetre. The vertical jump has been used to represent the core functional output of children's strength in previous literature [ 44]. It is also utilised readily in mainstream sport and physical education and is included in Australia's national talent identification and development program [ 39].

Functional Upper Limb Strength: Seated throw [39]

The seated throw measures functional upper limb strength. Children are seated comfortably with their back against a wall. Using a chest pass, they are asked to throw a basketball as far as possible while keeping their back against the wall. The distance between the wall and the first point of contact of the ball will be measured to the nearest centimetre for three throws and the average calculated. Similar to the vertical jump, the seated throw has been used to represent the core functional output of children's strength in previous literature [ 44, 45], is common and clinically feasible in mainstream sport and physical education and is included in Australia's national talent identification and development program [ 39].

Activity and Participation outcomes

Individual activity and participation based goals: Canadian Occupational Performance Measure (COPM) [46]

The COPM is the most frequently used measure of individual client centred outcomes in paediatric rehabilitation [ 47]. It measures individual, client-centred outcomes by focussing on the goals and priorities of the child and family [ 48]. The child-adapted model of the COPM will be administered via semi-structured interview with the parent/caregiver and child. Three sports related goals (at least one activity and one participation focussed) will be identified by caregivers. Ratings scale of their child's performance and their satisfaction with this performance will be made on a 1-10 ordinal scale. The COPM has high re-test reliability (ICC 0.76-0.89). It demonstrates concurrent validity with the Functional Independence Measure and Klein-Bell [ 49] in addition to construct and criterion validity [ 50]. It has good sensitivity to change [ 49]. On the ordinal scale (1-10) a change of two or more points is considered clinically meaningful [ 51].

Functional Mobility and Balance: Timed up and go (TUG) [52]

The TUG is a simple measure of balance, anticipatory postural control and functional mobility. The modified procedure for children described by Williams et al. [ 52] requires participants to stand up from a chair with a backrest but no arms, walk three meters to touch a target before turning and returning to a seated position. They are timed from their bottom rising from the seat to touching back down on the seat and are given encouragement throughout the procedure. The TUG has a high within-session and test-retest reliability (ICC 0.99) [ 53]. It is reported to be an ecologically valid tool. The TUG is responsive to change over time in children with physical disabilities [ 52]. Minimal detectable changes of 1.4 s (GMFCS- E&R I) and 2.87 s (GMFCS- E&R II) have been calculated [ 54].

Gross Motor Capacity (CP Specific): Gross Motor Function Measure Challenge Module (GMFM Challenge) [55]

The GMFM Challenge was developed as an extension of the GMFM which is used internationally to quantify gross motor performance in children with CP. The GMFM can have a ceiling effect, especially for children of GMFCS- E&R I classification over the age of 5 years. The GMFM Challenge is an observational measure of high-level skills, speed and quality of performance in children with ambulatory CP. It has been found to have excellent inter-rater (ICC = 0.97) and test-retest reliability (ICC = 0.96) [ 56]. The content validity of the GMFM Challenge was enhanced by using existing, feasible and relevant observational gross motor measures, working with experienced clinicians and performing participant based content validity checking with children with CP. Rasch analysis has been completed but not yet published [ 57]. Preliminary minimal detectable change values have been reported at 7.17-8.44 [ 56].

Gross Motor Capacity (Sport Specific): Test of Gross Motor Development-2 (TGMD-2) [58]

The TGMD-2 is an observational measure of gross motor skill performance relating to sports. It assesses skills in two categories, locomotor and object control, each with six items. In doing this, the TGMD-2 focusses on specific sports skills and is often used for children in mainstream education or sporting contexts and has been reported to be the gold standard for gross motor skill for pre-schoolers [ 59]. It is reported to have high inter-rater, test-retest, internal and composite reliability [ 58, 60, 61, 62]. Ulrich [ 58] reports excellent validity and clinical feasibility in the TGMD-2 manual and studies have demonstrated construct validity [ 61] and concurrent validity with the GMFCS- E&R [ 62], Pre-schooler Gross Motor Quality Scale [ 59] and measures of physical fitness [ 63].

Participation Frequency and Enjoyment: Children's Assessment of Participation and Enjoyment (CAPE) and Preferences of Activities for Children (PAC) [64]

The CAPE and PAC are questionnaires that measure participation of children in a range of activities outside of school. The CAPE-PAC measures who a child is participating with, enjoyment of an activity and the diversity and intensity of participation in formal (organised sport, other skill-based activities, and clubs, groups and organisations) and informal activities (recreational, active-physical, social, skill-based, and self-improvement). It has adequate test-retest reliability (ICC = 0.67-0.86) [ 65]. There is evidence for construct and face validity and clinical utility [ 65, 66].

Contextual

Quality of Life: Cerebral Palsy Quality of Life- children's version (CP QOL-Child) [67]

The CP QOL-Child is a quality of life questionnaire that assesses wellbeing in seven domains. It was specifically designed for children with CP aged 4-12 years. Parent proxy reports will be used in this study due to the age of most children anticipated in the Sports Stars group. The CP QOL-Child demonstrates high internal consistency (ICC 0.74-0.92) and test-retest reliability (ICC 0.76-0.89) for the parent proxy report. It demonstrates adequate construct validity relative construct validity with the Child Health Questionnaire, KIDSCREEN and GMFCS- E&R [ 67].

Caregiver satisfaction

Post intervention (T2) a custom-designed questionnaire will be used to measure satisfaction with the Sports Stars program. Questions will relate to group design, group content, and satisfaction with sport readiness. Outcomes will be measured on a Sports Stars specific eleven point Likert Scales with an additional open-ended question in each category. Answers will be reviewed by the primary investigator (GC) to determine consistent themes.

Child's motivation and engagement within the Sports Stars group

To evaluate if there is a relationship between study outcomes and participant engagement, treating physiotherapists will be asked to record participants' engagement in each component of the group intervention (warm-up, locomotor skills, object control skills, game participation and cool-down), in each therapy session using Sports Stars specific five point Likert Scales. Thematic analysis will be performed by to determine overarching themes.

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SPORTS STARS study protocol: a randomised, controlled trial of the effectiveness of a physiotherapist-led modified sport intervention for ambulant school-aged children with cerebral palsy

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Modified sport interventions run by physiotherapists have shown potential as cost-effective, engaging, and effective interventions to improve gross motor skills and support transition to real-world sports participation for children with cerebral palsy. At present, this population demonstrates decreased participation in physical activities and sport compared to peers due to barriers ranging from body function to accessibility challenges.

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Developed to update my host institution on progress of a state-wide clinical trial. Shared via email and intranet to state-wide allied health organisation.

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Developed to enhance the dissemination of my recently published protocol paper through social media. Measurable increase in altmetrics & metrics observed in the following week.