Test-retest reliability of corticokinematic coherence in young children with cerebral palsy: An observational longitudinal study.

OBJECTIVES: To assess the test-retest reliability of the corticokinematic coherence (CKC), an electrophysiological marker of proprioception, in children with cerebral palsy (CP). METHODS: Electroencephalography (EEG) signals from 15 children with unilateral or bilateral CP aged 23 to 53 months were recorded in two sessions 3 months apart using 128-channel EEG caps. During each session, children's fingers were moved at 2 Hz by an experimenter, in separate recordings for the more-affected (MA) and less-affected (LA) hands. The CKC was computed at the electrode and source levels, at movement frequency F0 (2 Hz) and its first harmonic F1 (4 Hz). A two-way mixed-effects model intraclass-correlation coefficient (ICC) was computed for the maximum CKC strength across electrodes at F0 and F1 obtained during the two sessions. RESULTS: ICC of the CKC strength acquired from LA and MA hands pooled together were respectively 0.51 (95% CI: 0.30-0.68) at F0 and 0.96 (95% CI: 0.93-0.98) at F1. The mean distances separating the CKC peaks in the source space at the two evaluation times were in the order of a centimeter. CONCLUSION: CKC is a robust electrophysiologic marker to study the longitudinal changes in cortical processing of proprioceptive afferences in young children with CP.

Bone Deformities through the Prism of the International Classification of Functioning, Disability and Health in Ambulant Children with Cerebral Palsy: A Systematic Review.

(1) Aim: The aim of this study was to determine the relationship between lower limb bone deformities and body functions, activity, and participation in ambulant children with CP and whether changing bone morphology affects outcomes in these domains. (2) Methods: A systematic literature search (PROSPERO CRD42020208416) of studies reporting correlations between measures of lower limb bone deformities and measures of body function, activity or participation, or post-surgical outcomes in these domains was conducted from 1990 to 2023 in Medline, Scopus, and Cochrane Library. We assessed study quality with the Checklist for Case Series (CCS) and a quality assessment developed by Quebec University Hospital. Meta-analysis was not possible; therefore, descriptive synthesis was performed. (3) Results: A total of 12 of 3373 screened articles were included. No studies evaluated the relationships between bone deformities and activity or participation, or the effect of isolated bone surgery on these domains. Correlations between bone deformities and body functions were poor-to-moderate. Internal hip rotation during gait improved after femoral derotation osteotomy. (4) Conclusions: A shift in paradigm is urgently required for the research and management of bone deformities in children with CP to include the activity and participation domains of the ICF, as well as consider more psychological aspects such as self-image.

Changes Induced by Early Hand-Arm Bimanual Intensive Therapy Including Lower Extremities in Young Children With Unilateral Cerebral Palsy: A Randomized Clinical Trial.

Importance: Intensive interventions are provided to young children with unilateral cerebral palsy (UCP), classically focused on the upper extremity despite the frequent impairment of gross motor function. Hand-Arm Bimanual Intensive Therapy Including Lower Extremities (HABIT-ILE) effectively improves manual dexterity and gross motor function in school-aged children. Objective: To verify if HABIT-ILE would improve manual abilities in young children with UCP more than usual motor activity. Design, Setting, and Participants: This prospective randomized clinical trial (November 2018 to December 2021), including 2 parallel groups and a 1:1 allocation, recruitment took place at European university hospitals, cerebral palsy specialized centers, and spontaneous applications at 3 sites: Brussels, Belgium; Brest, France; and Pisa, Italy. Matched (age at inclusion, lesion type, cause of cerebral palsy, and affected side) pairs randomization was performed. Young children were assessed at baseline (T0), 2 weeks after baseline (T1), and 3 months after baseline (T2). Health care professionals and assessors of main outcomes were blinded to group allocation. At least 23 young children (in each group) aged 12 to 59 months with spastic/dyskinetic UCP and able to follow instructions were needed. Exclusion criteria included uncontrolled seizures, scheduled botulinum toxin injections, orthopedic surgery scheduled during the 6 months before or during the study period, severe visual/cognitive impairments, or contraindications to magnetic resonance imaging. Interventions: Two weeks of usual motor activity including usual rehabilitation (control group) vs 2 weeks (50 hours) of HABIT-ILE (HABIT-ILE group). Main Outcomes and Measures: Primary outcome: Assisting Hand Assessment (AHA); secondary outcomes: Gross Motor Function Measure-66 (GMFM-66), Pediatric Evaluation of Disability Inventory-Computer Adaptive Test (PEDI-CAT), and Canadian Occupational Performance Measure (COPM). Results: Of 50 recruited young children (26 girls [52%], median age; 35.3 months for HABIT-ILE group; median age, 32.8 months for control group), 49 were included in the final analyses. Change in AHA score from T0 to T2 was significantly greater in the HABIT-ILE group (adjusted mean score difference [MD], 5.19; 95% CI, 2.84-7.55; P < .001). Changes in GMFM-66 (MD, 4.72; 95% CI, 2.66-6.78), PEDI-CAT daily activities (MD, 1.40; 95% CI, 0.29-2.51), COPM performance (MD, 3.62; 95% CI, 2.91-4.32), and satisfaction (MD, 3.53; 95% CI, 2.70-4.36) scores were greater in the HABIT ILE group. Conclusions and Relevance: In this clinical trial, early HABIT-ILE was shown to be an effective treatment to improve motor performance in young children with UCP. Moreover, the improvements had an impact on daily life activities of these children. Trial registration: ClinicalTrials.gov Identifier: NCT04020354.

Teaching and Rehabilitation of Handwriting for Children in the Digital Age: Issues and Challenges.

Handwriting is a determining factor for academic success and autonomy for all children. Making knowledge accessible to all is a challenge in the context of inclusive education. Given the neurodevelopmental diversity within a classroom of children, ensuring that the handwriting of all pupils progresses is very demanding for education professionals. The development of tools that can take into account the variability of the profiles and learning abilities of children with handwriting difficulties offers a new potential for the development of specific and adapted remediation strategies. This narrative review aims to present and discuss the challenges of handwriting learning and the opportunities offered by new technologies involving AI for school and health professionals to successfully improve the handwriting skills of all children.

Detecting Movement Changes in Children with Hemiparesis after Upper Limb Therapies: A Responsiveness Analysis of a 3D Bimanual Protocol.

The "Be an Airplane Pilot" (BE API) protocol was developed to evaluate upper limb (UL) kinematics in children with unilateral cerebral palsy (uCP) during bimanual tasks. The aim of this study was to investigate the responsiveness of this protocol to changes in kinematics and movement quality after UL therapies, using individual and group analyses, and to analyse the relationships between kinematic and functional changes in these children. Twenty children with uCP (5-15 years old) either participated in bimanual intensive therapy or received UL botulinum toxin injections. All the children performed the BE API protocol and functional assessments (Assisting Hand Assessment [AHA]) before and after the interventions. The individual analyses found kinematic changes in 100% of the children after therapy. The group analysis found significantly higher trunk and shoulder deviations after the intensive therapy. No significant changes were found for smoothness or trajectory straightness. The changes in the kinematic deviations were moderately correlated with the changes in the AHA scores. This study confirmed the responsiveness of the BE API protocol to change after therapy; therefore, the protocol is now fully validated and can be implemented in clinical practice. Its use should help in the accurate identification of impairments so that individualized treatments can be proposed.

Comprehensive personalized ankle joint shape analysis of children with cerebral palsy from pediatric MRI.

Cerebral palsy, a common physical disability in childhood, often causes abnormal patterns of movement and posture. To better understand the pathology and improve rehabilitation of patients, a comprehensive bone shape analysis approach is proposed in this article. First, a group analysis is performed on a clinical MRI dataset using two state-of-the-art shape analysis methods: ShapeWorks and a voxel-based method relying on Advanced Normalization Tools (ANTs) registration. Second, an analysis of three bones of the ankle is done to provide a complete view of the ankle joint. Third, a bone shape analysis is carried out at subject level to highlight variability patterns for personnalized understanding of deformities.

Assessing spino-cortical proprioceptive processing in childhood unilateral cerebral palsy with corticokinematic coherence.

OBJECTIVE: To develop an electrophysiological marker of proprioceptive spino-cortical tracts integrity based on corticokinematic coherence (CKC) in young children with unilateral cerebral palsy (UCP), in whom behavioral measures are not applicable. METHODS: Electroencephalography (EEG) signals from 12 children with UCP aged 19 to 57 months were recorded using 128-channel EEG caps while their fingers were moved at 2 Hz by an experimenter, in separate sessions for the affected and non-affected hands. The coherence between movement kinematics and EEG signals (i.e., CKC) was computed at the sensor and source (using a realistic head model) levels. Peaks of CKC obtained for the affected and non-affected hands were compared for location and strength. The relation between CKC strength on the lesion-side, the lesion-type (cortico-subcortical vs. subcortical) and the level of manual ability were studied with 2-way repeated-measures ANOVA. RESULTS: At the individual level, a significant CKC peak at the central area contralateral to the moved hand was found in all young children with their non-affected hand and in 8 out of 12 children with their affected hand. At the group level, CKC to the affected hand movements was weaker than CKC to the non-affected hand movements. This difference was influenced by the type of lesion, the effect being predominant in the subgroup (n = 5) with cortico-subcortical lesions. CONCLUSION: CKC is measurable with EEG in young children with UCP and provides electrophysiological evidence for altered proprioceptive spino-cortical tracts on the lesioned brain hemisphere, particularly in children with cortico-subcortical lesions.

Relationship between 3D lower limb bone morphology and 3D gait variables in children with uni and bilateral Cerebral Palsy.

BACKGROUND: Medical and surgical interventions to prevent or reduce bone deformities and improve gait in children with cerebral palsy (CP) are based on empirical evidence that there is a relationship between bone deformities and gait deviations. RESEARCH QUESTION: What is the relationship between tibial-femoral bone morphology and kinematic gait variables in ambulant children with CP? METHODS: A retrospective analysis was conducted on data from 121 children with uni- (n = 64, mean age 9.9 (SD 3.4) years) and bi- lateral (n = 57, mean age 10.4 (SD 3.6) years) CP who had undergone 3D gait analysis and biplanar X-rays (EOS® system). The limbs were split as DIP (the more impaired limb of children with bilateral CP), HEMI (the impaired limb of unilateral CP) and REF (the unimpaired limb of unilateral CP). Multi-variable Linear Regressions were performed between 23 kinematic variables, the Gait Deviation Index (GDI) and a model composed of nine 3D bone variables for each limb type. RESULTS: When the whole sample was pooled, 72% of R2 values were poor, 16% were fair, and 12% were moderate. Lower limb bone morphology models explained less than 1% of GDI variability. Correlations between tibial-femoral rotational parameters and hip rotation were mostly poor. Mean foot progression angle was the only kinematic parameter that was fairly to moderately correlated with bone variables in the 3 limb types. A tibial-femoral bone model explained 48% of the variability of mean foot progression angle in the REF limbs, 31% in the HEMI limbs and 25% in the DIP limbs. SIGNIFICANCE: Tibial-femoral bone morphology was only weakly related to kinematic gait variables, in contrast with common clinical assumptions. These results suggest that factors other than bone morphology influence gait quality and thus a thorough clinical examination and gait analysis is required prior to making treatment decisions.

Perceived impact of lockdown on daily life in children with physical disabilities and their families during the COVID-19 pandemic.

BACKGROUND: The first lockdown during COVID-19 pandemic in France led to an abrupt change in children's daily lives. For children with physical disabilities and their families, activities were limited, access to healthcare and therapy was disrupted, and family organization was altered. The objective was to report the impact of the lockdown on daily life activities and well-being of children with physical disabilities as perceived by caregivers. METHODS: Two online national surveys were addressed to the parents of children with physical disabilities (ECHO survey: 6 April to 11 May 2020) and without disabilities (E-COPAIN survey: 24 April to 11 May 2020), confined at home during the lockdown. A lockdown impact score was calculated from difficulties related to children's well-being (morale, behaviour and social interaction) and daily life activities (schooling and physical activity) and compared between groups. Data on family environment, parental stress and concerns were collected. RESULTS: One thousand three hundred seventy-six children (9.45 ± 4.78 years, 54% girls) in ECHO survey and 367 children (7.3 ± 4.4 years, 48% girls) in E-COPAIN survey were included. A negative impact of lockdown was found on 81% of children with physical disabilities. Behavioural problems were significantly more frequent (59.5% vs. 47.4%, P < .005) and parental stress was higher (6.1 ± 3.33 vs. 5.3 ± 3.01, P = .005) in the ECHO group. Associated impairments (odds ratio [OR] = 1.45 [1.30-1.62], P < .001), parental stress (OR = 1.09 [1.06-1.12], P < .001) and continuation of rehabilitation (OR = 0.80 [0.72-0.89], P < .001) were determinants of the level of difficulty experienced. CONCLUSIONS: The lockdown had a considerable, negative impact on the daily life of children with disabilities and their families. Guiding policymakers with the essential daily life activities and the services to provide for children with physical disabilities would offer valuable insights to manage such a sanitary crisis and allow to identify the most vulnerable population.

3-D lower extremity bone morphology in ambulant children with cerebral palsy and its relation to gait.

Changes in lower-extremity bone morphology are potential mid- to long-term secondary consequences of cerebral palsy (CP), affecting activity. Little is known about the 3-D morphology of lower-extremity bones in children with CP and the association with gait deviations. The main aim of this study was to describe and compare 3-D lower-extremity bone morphology in ambulant children with unilateral or bilateral CP. Secondary aims were to determine whether certain bone parameters were related to the unilateral or bilateral CP and to quantify the association between bone parameters and gait deviations. Among 105 ambulant children with CP (aged 3 to 17 years), 48 had bilateral CP (Bilat-CP) and 57 had unilateral CP (Unilat-CP); the unaffected limb of children with Unilat-CP was used as control limbs. Fifteen bone parameters were calculated by EOS® biplanar radiography, and the Gait Deviation Index (GDI) was calculated by 3-D gait analysis. Data were compared by descriptive and comparative statistical analysis (Anova, principal component analysis [PCA] and focused-PCA). Mean (SD) neck shaft angle was significantly greater for Unilat-CP than control limbs (134.9° [5.9] vs. 131.3° [5]). Mean mechanical tibial angle was significantly smaller (85.8° [6.7] vs. 89° [4.6]) and mean femoral torsion was significantly greater (29.4° [1.6] vs. 19.1° [11.8]) for Bilat-CP than control limbs. On PCA of the main determinants of 3-D bone morphology, bone shape was more complex with Bilat-CP, with changes in all 3 dimensions of space, than Unilat-CP and control limbs. Few bone parameters were correlated with the GDI in any limbs. In ambulant children with CP, femoral and tibial growth are not affected by the condition. The unilateral or bilateral nature of CP must be considered during treatment to prevent bone deformities and bone morphology affecting gait quality.

Emerging health challenges for children with physical disabilities and their parents during the COVID-19 pandemic: The ECHO French survey.

BACKGROUND: The daily lives of children with physical disabilities and their families have been significantly affected by the COVID-19 pandemic. The children face health risks, especially mental, behavioral, social and physical risks. OBJECTIVE: This study aimed to identify potential healthcare issues relating to the wellbeing of disabled children, continuity of rehabilitation and medical care, and parental concerns during the COVID-19 lockdown. METHODS: The Enfant Confinement Handicap besOins (ECHO [child lockdown disability needs]) national survey was developed by a multidisciplinary group and disseminated in France from April 6, 2020 via email and social networks. This online survey was addressed to the parents of children with physical disabilities aged 0 to 18 years. It explored the experiences of children and their families during the lockdown. Information regarding children's wellbeing, rehabilitation and family organization was collected. The first 1000 eligible surveys were analyzed. RESULTS: The children (mean [SD] age 9.5 [4.8] years) mostly had cerebral palsy (42%) or neuromuscular diseases (11%). The lockdown had negative effects on morale (44% of children), behaviour (55% of children) and social interactions (55% no contact with other children). Overall, 44% of children stopped physical activities; 76% were educated at home; 22% maintained medical follow-up, and 48% and 27% continued physiotherapy and occupational therapy respectively. For more than 60% of children, parents performed the therapy. The main parental concern was rehabilitation (72%) and their main difficulty was the mental load (50%); parents complained of lack of help and support (60%). CONCLUSIONS: This study highlighted substantial effects on the health of children with physical disabilities and loss of opportunity, with a massive interruption of medical follow-up and rehabilitation, during the lockdown. Regular assessment of the health benefit/risk is essential to support families and ensure continuity of care during a pandemic.

Protocol of changes induced by early Hand-Arm Bimanual Intensive Therapy Including Lower Extremities (e-HABIT-ILE) in pre-school children with bilateral cerebral palsy: a multisite randomized controlled trial.

BACKGROUND: Cerebral palsy (CP), which is the leading cause of motor disability during childhood, can produce sensory and cognitive impairments at different degrees. Most recent therapeutic interventions for these patients have solely focused on upper extremities (UE), although more than 60% of these patients present lower extremities (LE) deficits. Recently, a new therapeutic concept, Hand-arm Bimanual Intensive Therapy Including Lower Extremities (HABIT-ILE), has been proposed, involving the constant stimulation of UE and LE. Based on motor skill learning principles, HABIT-ILE is delivered in a day-camp setting, promoting voluntary movements for several hours per day during 10 consecutive week days. Interestingly, the effects of this intervention in a large scale of youngsters are yet to be observed. This is of interest due to the lack of knowledge on functional, neuroplastic and biomechanical changes in infants with bilateral CP. The aim of this randomized controlled study is to assess the effects of HABIT-ILE adapted for pre-school children with bilateral CP regarding functional, neuroplastic and biomechanical factors. METHODS: This international, multicentric study will include 50 pre-school children with CP from 12 to 60 months of age, comparing the effect of 50 h (2 weeks) of HABIT-ILE versus regular motor activity and/or customary rehabilitation. HABIT-ILE presents structured activities and functional tasks with continuous increase in difficulty while the child evolves. Assessments will be performed at 3 period times: baseline, two weeks later and 3 months later. The primary outcome will be the Gross Motor Function Measure 66. Secondary outcomes will include Both Hands Assessment, Melbourne Assessment-2, Semmes-Weinstein Monofilament Test, algometry assessments, executive function tests, ACTIVLIM-CP questionnaire, Pediatric Evaluation of Disability Inventory (computer adaptative test), Young Children's Participation and Environment Measure, Measure of the Process of Care, Canadian Occupational Performance Measure, neuroimaging and kinematics. DISCUSSION: The results of this study should highlight the impact of a motor, intensive, goal-directed therapy (HABIT-ILE) in pre-school children at a functional, neuroplastic and biomechanical level. In addition, this changes could demonstrated the impact of this intervention in the developmental curve of each child, improving functional ability, activity and participation in short-, mid- and long-term. NAME OF THE REGISTRY: Evaluation of Functional, Neuroplastic and Biomechanical Changes Induced by an Intensive, Playful Early-morning Treatment Including Lower Limbs (EARLY-HABIT-ILE) in Preschool Children With Uni and Bilateral Cerebral Palsy (HABIT-ILE). TRIAL REGISTRATION: NCT04017871 REGISTRATION DATE: July 12, 2019.