Maintenance of Functional Gains Following a Goal-Directed and FES-Assisted Cycling Program for Children With Cerebral Palsy.

PURPOSE: This study investigated whether the functional improvements associated with functional electrical stimulation-assisted cycling, goal-directed training, and adapted cycling in children with cerebral palsy were maintained 8 weeks after the intervention ceased. METHODS: The intervention (2 × 1-hour supervised sessions and 1-hour home program/week) ran for 8 weeks. Primary outcomes were the Gross Motor Function Measure (GMFM-88) and the Canadian Occupational Performance Measure (COPM). Secondary outcomes included the GMFM-66 and goal scores, 5 times sit-to-stand test (FTSTS), Participation and Environment Measure-Children and Youth (PEM-CY), Pediatric Evaluation of Disability Inventory Computer Adaptive Test (PEDI-CAT), and cycling power output (PO). Outcomes were assessed at baseline, 8 and 16 weeks. RESULTS: Twenty children participated (mean age = 10 years 3 months; SD = 2 years 11 months; Gross Motor Function Classification System II = 5, III = 6, and IV = 9). Improvements were retained above baseline at 16 weeks on the GMFM and COPM. Improvements in cycling PO, PEDI-CAT scores, PEM-CY environmental barriers and FTSTS were also retained. CONCLUSION: Functional improvements in children with cerebral palsy were retained 8 weeks post-intervention.

Functional electrical stimulation cycling, goal-directed training, and adapted cycling for children with cerebral palsy: a randomized controlled trial.

AIM: To test the efficacy of functional electrical stimulation (FES) cycling, goal-directed training, and adapted cycling, compared with usual care, to improve function in children with cerebral palsy (CP). METHOD: The intervention was delivered between 2017 and 2019 and included three sessions per week for 8 weeks (2×1h sessions at a children's hospital, and 1h home programme/week). Hospital sessions included 30 minutes of FES cycling and 30 minutes of goal-directed training. Home programmes included goal-directed training and adapted cycling. The comparison group continued usual care. Primary outcomes were gross motor function assessed by the Gross Motor Function Measure (GMFM) and goal performance/satisfaction assessed using the Canadian Occupational Performance Measure (COPM). Secondary outcomes were sit-to-stand and activity capacity, participation in home, school, and community activities, and power output. Linear regression was used to determine the between-group mean difference immediately post-training completion after adjusting for baseline scores. RESULTS: This randomized controlled trial included 21 participants (mean age=10y 3mo, standard deviation [SD]=3y; Gross Motor Function Classification System level: II=7, III=6, IV=8) who were randomized to the intervention (n=11) or usual care group (n=10). Between-group differences at T2 favoured the intervention group for GMFM-88 (mean difference=7.4; 95% confidence interval [CI]: 2.3-12.6; p=0.007), GMFM-66 (mean difference=5.9; 95% CI: 3.1-8.8; p<0.001), COPM performance (mean difference=4.4; 95% CI: 3.9-5.3; p<0.001) and satisfaction (mean difference=5.2; 95% CI: 4.0-6.4; p<0.001). INTERPRETATION: Children with CP achieved meaningful functional improvements after FES cycling, goal-directed training, and adapted cycling training. Cycling programmes for children with CP should be individualized and goal directed.

Efficacy of cycling interventions to improve function in children and adolescents with cerebral palsy: a systematic review and meta-analysis.

OBJECTIVES: The aim of this study was to determine the efficacy of cycling to improve function and reduce activity limitations in children with cerebral palsy; the optimal training parameters for improved function; and whether improvements in function can be retained. METHOD: Six databases were searched (until February 2019) and articles were screened in duplicate. Randomized or quasi-randomized controlled trials and pre-post studies were included. Methodological quality was assessed using the Downs and Black scale. Outcomes were reported under the International Classification of Functioning, Disability and Health domains of body functions and activity limitations. Quantitative analyses were completed using RevMan V5.3. RESULTS: A total of 533 articles were identified and 9 studies containing data on 282 participants met full inclusion criteria. Methodological quality ranged from low (14 of 32) to high (28 of 32). Significant improvements were reported for hamstring strength (effect size = 0.77-0.93), cardiorespiratory fitness (effect size = 1.13-1.77), balance (effect size = 1.03-1.29), 3-minute walk test distance (effect size = 1.14) and gross motor function (effect size = 0.91). Meta-analysis suggested that cycling can improve gross motor function (standardized mean difference = 0.35; 95% confidence interval = (-0.01, 0.70); P = 0.05); however, the effect was insignificant when a poor-quality study was omitted. CONCLUSION: Cycling can improve muscle strength, balance and gross motor function in children with cerebral palsy; however, optimal training doses are yet to be determined. There was insufficient data to determine whether functional improvements can be retained. Conclusions were limited by small sample sizes, inconsistent outcome measures and a lack of follow-up testing.

Australian hip surveillance guidelines for children with cerebral palsy: 5-year review.

AIM: To ensure hip surveillance guidelines reflect current evidence of factors influencing hip displacement in children with cerebral palsy (CP). METHOD: A three-step review process was undertaken: (1) systematic literature review, (2) analysis of hip surveillance databases, and (3) national survey of orthopaedic surgeons managing hip displacement in children with CP. RESULTS: Fifteen articles were included in the systematic review. Quantitative analysis was not possible. Qualitative review indicated hip surveillance programmes have decreased the incidence of hip dislocation in populations with CP. The Gross Motor Function Classification System was confirmed as the best indicator of risk for displacement, and evidence was found of hip displacement occurring at younger ages and in young adulthood. Femoral geometry, pelvic obliquity, and scoliosis were linked to progression of hip displacement. A combined data pool of 3366 children from Australian hip surveillance databases supported the effectiveness of the 2008 Consensus Statement to identify hip displacement early. The survey of orthopaedic surgeons supported findings of the systematic review and database analyses. INTERPRETATION: This review rationalized changes to the revised and renamed Australian Hip Surveillance Guidelines for Children with Cerebral Palsy 2014, informing frequency of radiographic examination in lower risk groups and continuation of surveillance into adulthood for adolescents with identified risk factors.

Botulinum toxin A for nonambulatory children with cerebral palsy: a double blind randomized controlled trial.

OBJECTIVES: To examine the efficacy and safety of intramuscular botulinum toxin A (BoNT-A) to reduce spasticity and improve comfort and ease of care in nonambulant children with cerebral palsy (CP). STUDY DESIGN: Nonambulant children with CP (n = 41; Gross Motor Function Classification System level IV = 3, level V = 38; mean age 7.1 years, range 2.3-16 years, 66% male) were randomly allocated to receive either intramuscular BoNT-A injections (n = 23) or sham procedure (n = 18) combined with therapy. The analysis used generalized estimating equations with primary outcome the Canadian Occupational Performance Measure (COPM) at 4 weeks postintervention and retention of effects at 16 weeks. Adverse events (AE) were collected at 2, 4, and 16 weeks by a physician masked to group allocation. RESULTS: There were significant between group differences favoring the BoNT-A-treated group on COPM performance at 4 weeks (estimated mean difference 2.2, 95% CI 0.8, 3.5; P = .002) and for COPM satisfaction (estimated mean difference 2.2, 95% CI 0.5, 3.9; P = .01). These effects were retained at 16 weeks for COPM satisfaction (estimated mean difference 1.8, 95% CI 0.1, 3.5; P = .04). There were more mild AE at 4 weeks for the BoNT-A group (P = .002), however, there were no significant between-group differences in the reporting of moderate and serious AE. CONCLUSIONS: In a double-blind randomized sham-controlled trial, intramuscular BoNT-A and therapy were effective for improving ease of care and comfort for nonambulant children with CP. There was no increase in moderate and severe AE in the children who had BoNT-A injections compared with the sham group.

GRIN: "GRoup versus INdividual physiotherapy following lower limb intra-muscular Botulinum Toxin-A injections for ambulant children with cerebral palsy: an assessor-masked randomised comparison trial": study protocol.

BACKGROUND: Cerebral palsy is the most common cause of physical disability in childhood. Spasticity is a significant contributor to the secondary impairments impacting functional performance and participation. The most common lower limb spasticity management is focal intramuscular injections of Botulinum Toxin-Type A accompanied by individually-delivered (one on one) physiotherapy rehabilitation. With increasing emphasis on improving goal-directed functional activity and participation within a family-centred framework, it is timely to explore whether physiotherapy provided in a group could achieve comparable outcomes, encouraging providers to offer flexible models of physiotherapy delivery. This study aims to compare individual to group-based physiotherapy following intramuscular Botulinum Toxin-A injections to the lower limbs for ambulant children with cerebral palsy aged four to fourteen years. METHODS/DESIGN: An assessor-masked, block randomised comparison trial will be conducted with random allocation to either group-based or individual physiotherapy. A sample size of 30 (15 in each study arm) will be recruited. Both groups will receive six hours of direct therapy following Botulinum Toxin-A injections in either an individual or group format with additional home programme activities (three exercises to be performed three times a week). Study groups will be compared at baseline (T1), then at 10 weeks (T2, efficacy) and 26 weeks (T3, retention) post Botulinum Toxin-A injections. Primary outcomes will be caregiver/s perception of and satisfaction with their child's occupational performance goals (Canadian Occupational Performance Measure) and quality of gait (Edinburgh Visual Gait Score) with a range of secondary outcomes across domains of the International Classification of Disability, Functioning and Health. DISCUSSION: This paper outlines the study protocol including theoretical basis, study hypotheses and outcome measures for this assessor-masked, randomised comparison trial comparing group versus individual models of physiotherapy following intramuscular injections of Botulinum Toxin-A to the lower limbs for ambulant children with cerebral palsy. TRIAL REGISTRATION: ACTRN12611000454976.

Relationship between early infant motor repertoire and neurodevelopment on the hammersmith infant neurological examination in a developmentally vulnerable First Nations cohort.

AIM: To implement a culturally-adapted screening program aimed to determine the ability of infant motor repertoire to predict early neurodevelopment on the Hammersmith Infant Neurological Examination (HINE) and improve Australian First Nations families' engagement with neonatal screening. METHODS: A prospective cohort of 156 infants (55 % male, mean (standard deviation [SD]) gestational age 33.8 (4.6) weeks) with early life risk factors for adverse neurodevelopmental outcomes (ad-NDO) participated in a culturally-adapted screening program. Infant motor repertoire was assessed using Motor Optimality Score-revised (MOS-R), captured over two videos, 11-13+6 weeks (V1; <14 weeks) and 14-18 weeks (V2; ≥14 weeks) corrected age (CA). At 4-9 months CA neurodevelopment was assessed on the HINE and classified according to age-specific cut-off and optimality scores as; developmentally 'on track' or high chance of either adverse neurodevelopmental outcome (ad-NDO) or cerebral palsy (CP). RESULTS: Families were highly engaged, 139/148 (94 %) eligible infants completing MOS-R, 136/150 (91 %), HINE and 123 (83 %) both. Lower MOS-R at V2 was associated with reduced HINE scores (ß = 1.73, 95 % confidence interval [CI] = 1.03-2.42) and high chance of CP (OR = 2.63, 95%CI = 1.21-5.69) or ad-NDO (OR = 1.38, 95%CI = 1.10-1.74). The MOS-R sub-category 'observed movement patterns' best predicted HINE, infants who score '4' had mean HINE 19.4 points higher than score '1' (95%CI = 12.0-26.9). Receiver-operator curve analyses determined a MOS-R cut-off of <23 was best for identifying mild to severely reduced HINE scores, with diagnostic accuracy 0.69 (sensitivity 0.86, 95%CI 0.76-0.94 and specificity 0.40, 95 % CI 0.25-0.57). A trajectory of improvement on MOS-R (≥2 point increase in MOS-R from 1st to 2nd video) significantly increased odds of scoring optimally on HINE (OR = 5.91, 95%CI 1.16-29.89) and may be a key biomarker of 'on track' development. INTERPRETATION: Implementation of a culturally-adapted program using evidence-based assessments demonstrates high retention. Infant motor repertoire is associated with HINE scores and the early neurodevelopmental status of developmentally vulnerable First Nations infants.

Australian Cerebral Palsy Child Study: protocol of a prospective population based study of motor and brain development of preschool aged children with cerebral palsy.

BACKGROUND: Cerebral palsy (CP) results from a static brain lesion during pregnancy or early life and remains the most common cause of physical disability in children (1 in 500). While the brain lesion is static, the physical manifestations and medical issues may progress resulting in altered motor patterns. To date, there are no prospective longitudinal studies of CP that follow a birth cohort to track early gross and fine motor development and use Magnetic Resonance Imaging (MRI) to determine the anatomical pattern and likely timing of the brain lesion. Existing studies do not consider treatment costs and outcomes. This study aims to determine the pathway(s) to motor outcome from diagnosis at 18 months corrected age (c.a.) to outcome at 5 years in relation to the nature of the brain lesion (using structural MRI). METHODS: This prospective cohort study aims to recruit a total of 240 children diagnosed with CP born in Victoria (birth years 2004 and 2005) and Queensland (birth years 2006-2009). Children can enter the study at any time between 18 months to 5 years of age and will be assessed at 18, 24, 30, 36, 48 and 60 months c.a. Outcomes include gross motor function (GMFM-66 & GMFM-88), Gross Motor Function Classification System (GMFCS); musculoskeletal development (hip displacement, spasticity, muscle contracture), upper limb function (Manual Ability Classification System), communication difficulties using Communication and Symbolic Behaviour Scales-Developmental Profile (CSBS-DP), participation using the Paediatric Evaluation of Disability Inventory (PEDI), parent reported quality of life and classification of medical and allied health resource use and determination of the aetiology of CP using clinical evaluation combined with MRI. The relationship between the pathways to motor outcome and the nature of the brain lesion will be analysed using multiple methods including non-linear modelling, multilevel mixed-effects models and generalised estimating equations. DISCUSSION: This protocol describes a large population-based study of early motor development and brain structure in a representative sample of preschool aged children with CP, using direct clinical assessment. The results of this study will be published in peer reviewed journals and presented at relevant international conferences. TRIAL REGISTRATION: Australia and New Zealand Clinical Trials Register (ACTRN1261200169820).

ACTIVE STRIDES-CP: protocol for a randomised trial of intensive rehabilitation (combined intensive gait and cycling training) for children with moderate-to-severe bilateral cerebral palsy.

INTRODUCTION: For children with cerebral palsy (CP), who are marginally ambulant, gross motor capacity peaks between 6 and 7 years of age with a subsequent clinical decline, impacting their ability to engage in physical activity. Active Strides-CP is a novel package of physiotherapy targeting body functions, activity and participation outcomes for children with bilateral CP. This study will compare Active Strides-CP to usual care in a multisite randomised waitlist-controlled trial. METHODS AND ANALYSIS: 150 children with bilateral CP (5-15 years), classified in Gross Motor Function Classification System (GMFCS) levels III and IV will be stratified (GMFCS III vs IV, age 5-10 years; 11-15 years and trial site) and randomised to receive either (1) 8 weeks of Active Strides-CP two times/week for 1.5 hours in clinic and one time/week for 1 hour alternating home visits and telehealth (total dose=32 hours) or (2) usual care. Active Strides-CP comprises functional electrical stimulation cycling, partial body weight support treadmill training, overground walking, adapted community cycling and goal-directed training. Outcomes will be measured at baseline, immediately post-intervention at 9 weeks primary endpoint and at 26 weeks post-baseline for retention. The primary outcome is the Gross Motor Function Measure-66. Secondary outcomes include habitual physical activity, cardiorespiratory fitness, walking speed and distance, frequency/involvement of community participation, mobility, goal attainment and quality of life. Analyses will follow standard principles for randomised controlled trials using two-group comparisons on all participants on an intention-to-treat basis. Comparisons between groups for primary and secondary outcomes will be conducted using regression models. A within-trial cost utility analysis will be performed. ETHICS AND DISSEMINATION: The Children's Health Queensland Hospital and Health Service, The University of Queensland, The University of Melbourne and Curtin University Human Research Ethics Committees have approved this study. Results will be disseminated as conference abstracts and presentations, peer-reviewed articles in scientific journals, and institution newsletters and media releases. TRIAL REGISTRATION NUMBER: ACTRN12621001133820.