Lateralized modulation of cortical beta power during human gait is related to arm swing.

Human gait is a complex behavior requiring dynamic control of upper and lower extremities that is accompanied by cortical activity in multiple brain areas. We investigated the contribution of beta (15-30 Hz) and gamma (30-50 Hz) band electroencephalography (EEG) activity during specific phases of the gait cycle, comparing treadmill walking with and without arm swing. Modulations of spectral power in the beta band during early double support and swing phases source-localized to the sensorimotor cortex ipsilateral, but not contralateral, to the leading leg. The lateralization disappeared in the condition with constrained arms, together with an increase of activity in bilateral supplementary motor areas. By contrast, gamma band modulations that localized to the presumed leg area of sensorimotor cortex around the heel-strike events were unaffected by arm movement. Our findings demonstrate that arm swing is accompanied by considerable cortical activation that should not be neglected in gait-related neuroimaging studies.

Motor function and gait decline in individuals with cerebral palsy during adulthood: a narrative review of potential physiological determinants.

Cerebral palsy (CP) is the most common childhood-onset disability. The evolution of gait according to severity is well known amongst children and thought to peak between 8 and 12 years of age among those walking without assistive devices. However, among adults, clinical experience as well as scientific studies report, through clinical assessments, questionnaires and interviews, increasing walking difficulties leading to an increased dependency of assistive devices in everyday ambulation. For many individuals with CP, this change will occur around 30-40 years, with the risk of losing mobility increasing with age. This narrative review aims to first provide objective evidence of motor function and gait decline in adults with CP when ageing, and then to offer mechanistic hypotheses to explain those alterations. Many studies have compared individuals with CP to the typically developing population, yet the evolution with ageing has largely been understudied. Comorbid diagnoses comprise one of the potential determinants of motor function and gait decline with ageing in people with CP, with the first manifestations happening at an early age and worsening with ageing. Similarly, ageing appears to cause alterations to the neuromuscular and cardiovascular systems at an earlier age than their typically developing (TD) peers. Future studies should, however, try to better understand how the physiological particularities of CP change with ageing that could pave the way for better strategies for maintaining function and quality of life in people with CP.

Markerless motion analysis to assess reaching-sideways in individuals with dyskinetic cerebral palsy: A validity study.

This study aimed to evaluate clinical utility of 2D-markerless motion analysis (2DMMA) from a single camera during a reaching-sideways-task in individuals with dyskinetic cerebral palsy (DCP) by determining (1) concurrent validity by correlating 2DMMA against marker-based 3D-motion analysis (3DMA) and (2) construct validity by assessing differences in 2DMMA features between DCP and typically developing (TD) peers. 2DMMA key points were tracked from frontal videos of a single camera by DeepLabCut and accuracy was assessed against human labelling. Shoulder, elbow and wrist angles were calculated from 2DMMA and 3DMA (as gold standard) and correlated to assess concurrent validity. Additionally, execution time and variability features such as mean point-wise standard deviation of the angular trajectories (i.e. shoulder elevation, elbow and wrist flexion/extension) and wrist trajectory deviation by mean overshoot and convex hull were calculated from key points. 2DMMA features were compared between the DCP group and TD peers to assess construct validity. Fifty-one individuals (30 DCP;21 TD; age:5-24 years) participated. An accuracy of approximately 1.5 cm was reached for key point tracking. While significant correlations were found for wrist (ρ = 0.810;p < 0.001) and elbow angles (ρ = 0.483;p < 0.001), 2DMMA shoulder angles were not correlated (ρ = 0.247;p = 0.102) to 3DMA. Wrist and elbow angles, execution time and variability features all differed between groups (Effect sizes 0.35-0.81;p < 0.05). Videos of a reaching-sideways-task processed by 2DMMA to assess upper extremity movements in DCP showed promising validity. The method is especially valuable to assess movement variability.

A Novel Video-Based Methodology for Automated Classification of Dystonia and Choreoathetosis in Dyskinetic Cerebral Palsy During a Lower Extremity Task.

BACKGROUND: Movement disorders in children and adolescents with dyskinetic cerebral palsy (CP) are commonly assessed from video recordings, however scoring is time-consuming and expert knowledge is required for an appropriate assessment. OBJECTIVE: To explore a machine learning approach for automated classification of amplitude and duration of distal leg dystonia and choreoathetosis within short video sequences. METHODS: Available videos of a heel-toe tapping task were preprocessed to optimize key point extraction using markerless motion analysis. Postprocessed key point data were passed to a time series classification ensemble algorithm to classify dystonia and choreoathetosis duration and amplitude classes (scores 0, 1, 2, 3, and 4), respectively. As ground truth clinical scoring of dystonia and choreoathetosis by the Dyskinesia Impairment Scale was used. Multiclass performance metrics as well as metrics for summarized scores: absence (score 0) and presence (score 1-4) were determined. RESULTS: Thirty-three participants were included: 29 with dyskinetic CP and 4 typically developing, age 14 years:6 months ± 5 years:15 months. The multiclass accuracy results for dystonia were 77% for duration and 68% for amplitude; for choreoathetosis 30% for duration and 38% for amplitude. The metrics for score 0 versus score 1 to 4 revealed an accuracy of 81% for dystonia duration, 77% for dystonia amplitude, 53% for choreoathetosis duration and amplitude. CONCLUSIONS: This methodology study yielded encouraging results in distinguishing between presence and absence of dystonia, but not for choreoathetosis. A larger dataset is required for models to accurately represent distinct classes/scores. This study presents a novel methodology of automated assessment of movement disorders solely from video data.

The applicability of markerless motion capture for clinical gait analysis in children with cerebral palsy.

The aim of this comparative, cross-sectional study was to determine whether markerless motion capture can track deviating gait patterns in children with cerebral palsy (CP) to a similar extent as marker-based motion capturing. Clinical gait analysis (CGA) was performed for 30 children with spastic CP and 15 typically developing (TD) children. Marker data were processed with the Human Body Model and video files with Theia3D markerless software, to calculate joint angles for both systems. Statistical parametric mapping paired t-tests were used to compare the trunk, pelvis, hip, knee and ankle joint angles, for both TD and CP, as well as for the deviation from the norm in the CP group. Individual differences were quantified using mean absolute differences. Markerless motion capture was able to track frontal plane angles and sagittal plane knee and ankle angles well, but individual deviations in pelvic tilt and transverse hip rotation as present in CP were not captured by the system. Markerless motion capture is a promising new method for CGA in children with CP, but requires improvement to better capture several clinically relevant deviations especially in pelvic tilt and transverse hip rotation.

Potentially Life-Threatening Interaction between Opioids and Intrathecal Baclofen in Individuals with a Childhood-Onset Neurological Disorder: A Case Series and Review of the Literature.

BACKGROUND: Spasticity and dystonia are movement impairments that can occur in childhood-onset neurological disorders. Severely affected individuals can be treated with intrathecal baclofen (ITB). Concomitant use of ITB and opioids has been associated with central nervous system (CNS) depression. This study aims to describe the clinical management of this interaction, based on a case series and review of literature. METHODS: Four individuals with childhood-onset CNS disorders (age 8-24) and CNS-depressant overdose symptoms after the concomitant use of ITB and opioids are described. The Drug Interaction Probability Scale (DIPS) was calculated to assess the cause-relationship (doubtful <2, possible 2-4, probable 5-8, and highly probable >8) of the potential drug-drug interaction. A literature review of similar previously reported cases and the possible pharmacological mechanisms of opioid-baclofen interaction is provided. RESULTS: After ITB and opioid co-administration, three out of four patients had decreased consciousness, and three developed respiratory depression. DIPS scores indicated a possible cause-relationship in one patient (DIPS: 4) and a probable cause-relationship in the others (DIPS: 6, 6, and 8). Discontinuation or adjusting ITB or opioid dosages resulted in clinical recovery. All patients recovered completely. In the literature, two articles describing nine unique cases were found. CONCLUSION: Although the opioid-ITB interaction is incompletely understood, concomitant use may enhance the risk of symptoms of CNS-depressant overdose, which are potentially life-threatening. If concomitant use is desirable, we strongly recommend to closely monitor these patients to detect interaction symptoms early. Awareness and monitoring of the potential opioid-ITB interaction is essential to reduce the risk of severe complications.

Exploring harmonic walking development in children with unilateral cerebral palsy and typically developing toddlers: Insights from walking experience.

This longitudinal study investigated the impact of the first independent steps on harmonic gait development in unilateral cerebral palsy (CP) and typically developing (TD) children. We analysed the gait ratio values (GR) by comparing the duration of stride/stance, stance/swing and swing/double support phases. Our investigation focused on identifying a potential trend towards the golden ratio value of 1.618, which has been observed in the locomotion of healthy adults as a characteristic of harmonic walking. Locomotor ability was assessed in both groups at different developmental stages: before and after the emergence of independent walking. Results revealed that an exponential fit was observed only after the first unsupported steps were taken. TD children achieved harmonic walking within a relatively short period (approximately one month) compared to children with CP, who took about seven months to develop harmonic walking. Converging values for stride/stance and stance/swing gait ratios, averaged on the two legs, closely approached the golden ratio in TD children (R2 = 0.9) with no difference in the analysis of the left vs right leg separately. In contrast, children with CP exhibited a trend for stride/stance and stance/swing (R2 = 0.7), with distinct trends observed for the most affected leg which did not reach the golden ratio value for the stride/stance ratio (GR = 1.5), while the least affected leg exceeded it (GR = 1.7). On the contrary, the opposite trend was observed for the stance/swing ratio. These findings indicate an overall harmonic walking in children with CP despite the presence of asymmetry between the two legs. These results underscore the crucial role of the first independent steps in the progressive development of harmonic gait over time.

The relationship between manual ability, dystonia and choreoathetosis severity and upper limb movement patterns during reaching and grasping in children and young adults with dyskinetic cerebral palsy.

INTRODUCTION: Impaired upper limb movements are a key feature in dyskinetic cerebral palsy (CP). However, information on how specific movement patterns relate to manual ability, performance and underlying movement disorders is lacking. Insight in these associations may contribute to targeted upper limb management in dyskinetic CP. This study aimed to explore associations between deviant upper limb movement patterns and (1) manual ability, (2) severity of dystonia/choreoathetosis, and (3) movement time/trajectory deviation during reaching and grasping. PARTICIPANTS/METHODS: Participants underwent three-dimensional upper limb analysis during reaching forwards (RF), reaching sideways (RS) and reach-and-grasp vertical (RGV) as well as clinical assessment. Canonical correlation and regression analysis with statistical parametric mapping were used to explore associations between clinical/performance parameters and movement patterns (mean and variability). RESULTS: Thirty individuals with dyskinetic CP participated (mean age 16±5 y; 20 girls). Lower manual ability was related to higher variability in wrist flexion/extension during RF and RS early in the reaching cycle (p < 0.05). Higher dystonia severity was associated with higher mean wrist flexion (40-82 % of the reaching cycle; p = 0.004) and higher variability in wrist flexion/extension (31-75 %; p < 0.001) and deviation (2-14 %; p = 0.007/60-73 %; p = 0.006) during RF. Choreoathetosis severity was associated with higher elbow pro/supination variability (12-19 %; p = 0.009) during RGV. Trajectory deviation was associated with wrist and elbow movement variability (p < 0.05). CONCLUSION: Current novel analysis of upper limb movement patterns and respective timings allows to detect joint angles and periods in the movement cycle wherein associations with clinical parameters occur. These associations are not present at each joint level, nor during the full movement cycle. This knowledge should be considered for individualized treatment strategies.

Multi-segment foot kinematics during gait in children with spastic cerebral palsy.

BACKGROUND: Foot deformities (e.g. planovalgus and cavovarus) are very common in children with spastic cerebral palsy (CP), with the midfoot often being involved. Dynamic foot function can be assessed with 3D gait analysis including a multi-segment foot model. Incorporating a midfoot segment in such a model, allows quantification of separate Chopart and Lisfranc joint kinematics. Yet, midfoot kinematics have not previously been reported in CP. RESEARCH QUESTIONS: What is the difference in multi-segment kinematics including midfoot joints between common foot deformities in CP and typically-developing feet? METHODS: 103 feet of 57 children with spastic CP and related conditions were retrospectively included and compared with 15 typically-developing children. All children underwent clinical gait analysis with the Amsterdam Foot Model marker set. Multi-segment foot kinematics were calculated for three strides per foot and averaged. A k-means cluster analysis was performed to identify foot deformity groups that were present within CP data. The deformity type represented by each cluster was based on the foot posture index. Kinematic output of the clusters was compared to typically-developing data for a static standing trial and for the range of motion and kinematic waveforms during walking, using regular and SPM independent t-tests respectively. RESULTS: A neutral, planovalgus and varus cluster were identified. Neutral feet showed mostly similar kinematics as typically-developing data. Planovalgus feet showed increased ankle valgus and Chopart dorsiflexion, eversion and abduction. Varus feet showed increased ankle varus and Chopart inversion and adduction. SIGNIFICANCE: This study is the first to describe Chopart and Lisfranc joint kinematics in different foot deformities of children with CP. It shows that adding a midfoot segment can provide additional clinical and kinematic information. It highlights joint angles that are more distinctive between deformities, which could be helpful to optimize the use of multi-segment foot kinematics in the clinical decision making process.

Physical fitness in children with Marfan and Loeys-Dietz syndrome: associations between cardiovascular parameters, systemic manifestations, fatigue, and pain.

Children with Marfan (MFS) and Loeys-Dietz syndrome (LDS) report limitations in physical activities, sports, school, leisure, and work participation in daily life. This observational, cross-sectional, multicenter study explores associations between physical fitness and cardiovascular parameters, systemic manifestations, fatigue, and pain in children with MFS and LDS. Forty-two participants, aged 6-18 years (mean (SD) 11.5(3.7)), diagnosed with MFS (n = 36) or LDS (n = 6), were enrolled. Physical fitness was evaluated using the Fitkids Treadmill Test's time to exhaustion (TTE) outcome measure. Cardiovascular parameters (e.g., echocardiographic parameters, aortic surgery, cardiovascular medication) and systemic manifestations (systemic score of the revised Ghent criteria) were collected. Pain was obtained by visual analog scale. Fatigue was evaluated by PROMIS® Fatigue-10a-Pediatric-v2.0-short-form and PROMIS® Fatigue-10a-Parent-Proxy-v2.0-short-form. Multivariate linear regression analyses explored associations between physical fitness (dependent variable) and independent variables that emerged from the univariate linear regression analyses (criterion p < .05). The total group (MFS and LDS) and the MFS subgroup scored below norms on physical fitness TTE Z-score (mean (SD) -3.1 (2.9); -3.0 (3.0), respectively). Univariate analyses showed associations between TTE Z-score aortic surgery, fatigue, and pain (criterion p < .05). Multivariate analyses showed an association between physical fitness and pediatric self-reported fatigue that explained 48%; 49%, respectively, of TTE Z-score variance (F (1,18) = 18.6, p ≤ .001, r2 = .48; F (1,15) = 16,3, p = .01, r2 = .49, respectively).    Conclusions: Physical fitness is low in children with MFS or LDS and associated with self-reported fatigue. Our findings emphasize the potential of standardized and tailored exercise programs to improve physical fitness and reduce fatigue, ultimately enhancing the physical activity and sports, school, leisure, and work participation of children with MFS and LDS. What is Known: • Marfan and Loeys-Dietz syndrome are heritable connective tissue disorders and share cardiovascular and systemic manifestations. • Children with Marfan and Loeys-Dietz syndrome report increased levels of disability, fatigue and pain, as well as reduced levels of physical activity, overall health and health-related quality of life. What is New: • Physical fitness is low in children with Marfan and Loeys-Dietz syndrome and associated with self-reported fatigue. • Our findings emphasize the potential of standardized and tailored exercise programs to improve physical fitness and reduce fatigue, ultimately enhancing the physical activity and sports, school, leisure, and work participation of children with Marfan and Loeys-Dietz syndrome.

Surgical treatment options for spasticity in children and adolescents with hereditary spastic paraplegia.

PURPOSE: To provide an overview of outcome and complications of selective dorsal rhizotomy (SDR) and intrathecal baclofen pump implantation (ITB) for spasticity treatment in children with hereditary spastic paraplegia (HSP). METHODS: Retrospective study including children with HSP and SDR or ITB. Gross motor function measure (GMFM-66) scores and level of spasticity were assessed. RESULTS: Ten patients were included (most had mutations in ATL1 (n = 4) or SPAST (n = 3) genes). Four walked without and two with walking aids, four were non-walking children. Six patients underwent SDR, three patients ITB, and one both. Mean age at surgery was 8.9 ± 4.5 years with a mean follow-up of 3.4 ± 2.2 years. Five of the SDR patients were walking. Postoperatively spasticity in the legs was reduced in all patients. The change in GMFM-66 score was + 8.0 (0-19.7 min-max). The three ITB patients treated (SPAST (n = 2) and PNPLA6 (n = 1) gene mutation) were children with a progressive disease course. No complications of surgery occurred. CONCLUSIONS: SDR is a feasible treatment option in carefully selected children with HSP, especially in walking patients. The majority of patients benefit with respect to gross motor function, complication risk is low. ITB was used in children with severe and progressive disease.

Child-to-adult transition: a survey of current practices within the European Reference Network for Rare Neurological Diseases (ERN-RND).

BACKGROUND: Transition from child-centered to adult-centered healthcare is a gradual process that addresses the medical, psychological, and educational needs of young people in the management of their autonomy in making decisions about their health and their future clinical assistance. This transfer is challenging across all chronic diseases but can be particularly arduous in rare neurological conditions. AIM: To describe the current practice on the transition process for young patients in centers participating in the European Reference Network for Rare Neurological Diseases (ERN-RND). METHODS: Members of the ERN-RND working group developed a questionnaire considering child-to-adult transition issues and procedures in current clinical practice. The questionnaire included 20 questions and was sent to members of the health care providers (HCPs) participating in the network. RESULTS: Twenty ERN-RND members (75% adult neurologists; 25% pediatricians; 5% nurses or study coordinators) responded to the survey, representing 10 European countries. Transition usually occurs between 16 and 18 years of age, but 55% of pediatric HCPs continue to care for their patients until they reach 40 years of age or older. In 5/20 ERN-RND centers, a standardized procedure managing transition is currently adopted, whereas in the remaining centers, the transition from youth to adult service is usually assisted by pediatricians as part of their clinical practice. CONCLUSIONS: This survey demonstrated significant variations in clinical practice between different centers within the ERN-RND network. It provided valuable data on existing transition programs and highlighted key challenges in managing transitions for patients with rare neurological disorders.

Predictive simulations identify potential neuromuscular contributors to idiopathic toe walking.

BACKGROUND: Most cases of toe walking in children are idiopathic. We used pathology-specific neuromusculoskeletal predictive simulations to identify potential underlying neural and muscular mechanisms contributing to idiopathic toe walking. METHODS: A musculotendon contracture was added to the ankle plantarflexors of a generic musculoskeletal model to represent a pathology-specific contracture model, matching the reduced ankle dorsiflexion range-of-motion in a cohort of children with idiopathic toe walking. This model was employed in a forward dynamic simulation controlled by reflexes and supraspinal drive, governed by a multi-objective cost function to predict gait patterns with the contracture model. We validated the predicted gait using experimental gait data from children with idiopathic toe walking with ankle contracture, by calculating the root mean square errors averaged over all biomechanical variables. FINDINGS: A predictive simulation with the pathology-specific model with contracture approached experimental ITW data (root mean square error = 1.37SD). Gastrocnemius activation was doubled from typical gait simulations, but lacked a peak in early stance as present in electromyography. This synthesised idiopathic toe walking was more costly for all cost function criteria than typical gait simulation. Also, it employed a different neural control strategy, with increased length- and velocity-based reflex gains to the plantarflexors in early stance and swing than typical gait simulations. INTERPRETATION: The simulations provide insights into how a musculotendon contracture combined with altered neural control could contribute to idiopathic toe walking. Insights into these neuromuscular mechanisms could guide future computational and experimental studies to gain improved insight into the cause of idiopathic toe walking.

Altered upper limb kinematics in individuals with dyskinetic cerebral palsy in comparison with typically developing peers - A statistical parametric mapping study.

BACKGROUND: Dyskinetic cerebral palsy (DCP) is clinically characterized by involuntary movements and abnormal postures, which can aggravate with activity. While upper limb movement variability is often detected in the clinical picture, it remains unknown how movement patterns of individuals with DCP differ from typically developing (TD) peers. RESEARCH QUESTION: Do individuals with DCP show i) higher time-dependent standard deviations of upper limb joint angles and ii) altered upper limb kinematics in time and/or amplitude during functional upper limb tasks in comparison with TD individuals? METHODS: Three-dimensional upper limb movement patterns were cross-sectionally compared in 50 individuals with and without DCP during three functional tasks: reach forward (RF), reach and grasp vertical (RGV) and reach sideways (RS). Mean and point-wise standard deviations of angular waveform of the upper limb joint angles were compared between groups to evaluate differences in time and/or amplitude using traditional and non-linear registration statistical parametric mapping. RESULTS: Thirty-five extremities from 30 individuals (mean age 17y4m, range 5-25 y; MACS level I(n = 2); II(n = 15); III(n = 16); IV(n = 2)) with DCP and twenty TD individuals (mean age 16y8m, range 8-25 y) were evaluated. The DCP compared to TD group showed higher point-wise standard deviations at the level of all joints, which was time-dependent and varied between tasks. Mean wrist and elbow flexion was higher for the DCP group during RF (0-83 % wrist; 57-100 % elbow), RGV (0-82 % wrist; 12-100 % elbow) and RS (0-43 % wrist; 70-100 % elbow). SIGNIFICANCE: This is the first study exploring the movement patterns of individuals with DCP during reaching using quantitative measures. Analyzing these individual movement patterns by statistical parametric mapping (SPM) allows us to focus on both specific joint or on specific timing during the movement cycle. The individual information that this method yields can guide individual therapy aiming to improve reaching function in different parts of the movement cycle or evaluate intervention effects on upper extremity treatment.

Predicting Gait Patterns of Children With Spasticity by Simulating Hyperreflexia.

Spasticity is a common impairment within pediatric neuromusculoskeletal disorders. How spasticity contributes to gait deviations is important for treatment selection. Our aim was to evaluate the pathophysiological mechanisms underlying gait deviations seen in children with spasticity, using predictive simulations. A cluster analysis was performed to extract distinct gait patterns from experimental gait data of 17 children with spasticity to be used as comparative validation data. A forward dynamic simulation framework was employed to predict gait with either velocity- or force-based hyperreflexia. This framework entailed a generic musculoskeletal model controlled by reflexes and supraspinal drive, governed by a multiobjective cost function. Hyperreflexia values were optimized to enable the simulated gait to best match experimental gait patterns. Three experimental gait patterns were extracted: (1) increased knee flexion, (2) increased ankle plantar flexion, and (3) increased knee flexion and ankle plantar flexion when compared with typical gait. Overall, velocity-based hyperreflexia outperformed force-based hyperreflexia. The first gait pattern could mostly be explained by rectus femoris and hamstrings velocity-based hyperreflexia, the second by gastrocnemius velocity-based hyperreflexia, and the third by gastrocnemius, soleus, and hamstrings velocity-based hyperreflexia. This study shows how velocity-based hyperreflexia from specific muscles contributes to different spastic gait patterns, which may help in providing targeted treatment.

Fatigue-related gait adaptations in children with cerebral palsy.

AIM: To obtain insights into the effects of fatigue on the kinematics, kinetics, and energy cost of walking (ECoW) in children with cerebral palsy (CP). METHOD: In this prospective observational study, 12 children with CP (mean age 12 years 9 months, SD 2 years 7 months; four females, eight males) and 15 typically developing children (mean age 10 years 8 months, SD 2 years 4 months; seven females, eight males) followed a prolonged intensity-based walking protocol on an instrumented treadmill, combined with gas analysis measurements. The protocol consisted of consecutive stages, including a 6-minute walking exercise (6MW) at comfortable speed, 2 minutes of moderate-intensity walking (MIW) (with a heart rate > 70% of its predicted maximal), and 4 minutes walking after MIW. If necessary, the speed and slope were incremented to reach MIW. Outcomes were evaluated at the beginning and end of the 6MW and after MIW. RESULTS: With prolonged walking, Gait Profile Scores deteriorated slightly for both groups (p < 0.01). Knee flexion increased during early stance (p = 0.004) and ankle dorsiflexion increased during late stance (p = 0.034) in children with CP only. Negligible effects were found for kinetics. No demonstrable change in ECoW was found in either group (p = 0.195). INTERPRETATION: Kinematic deviations in children with CP are progressive with prolonged walking. The large variation in adaptations indicates that an individual approach is recommended to investigate the effects of physical fatigue on gait in clinical practice.

Electromyographic biofeedback-driven gaming to alter calf muscle activation during gait in children with spastic cerebral palsy.

BACKGROUND: Children with cerebral palsy often show deviating calf muscle activation patterns during gait, with excess activation during early stance and insufficient activation during push-off. RESEARCH QUESTION: Can children with cerebral palsy improve their calf muscle activation patterns during gait using one session of biofeedback-driven gaming? METHODS: Eighteen children (6-17 y) with spastic cerebral palsy received implicit game-based biofeedback on electromyographic activity of the calf muscle (soleus or gastrocnemius medialis) while walking on a treadmill during one session. Biofeedback alternately aimed to reduce early stance activity, increase push-off activity, and both combined. Early stance and push-off activity and the double-bump-index (early stance divided by push-off activity) were determined during baseline and walking with feedback. Changes were assessed at group level using repeated measures ANOVA with simple contrast or Friedman test with post-hoc Wilcoxon signed rank test, as well as individually using independent t-tests or Wilcoxon rank sum tests. Perceived competence and interest-enjoyment were assessed through a questionnaire. RESULTS: Children successfully decreased their electromyographic activity during early stance feedback trials (relative decrease of 6.8 ± 12.2 %, P = 0.025), with a trend during the combined feedback trials (6.5 ± 13.9 %, P = 0.055), and increased their electromyographic activity during push-off feedback trials (8.1 ± 15.8 %, P = 0.038). Individual improvements were seen in twelve of eighteen participants. All children experienced high levels of interest-enjoyment (8.4/10) and perceived competence (8.1/10). SIGNIFICANCE: This exploratory study suggests that children with cerebral palsy can achieve small within-session improvements of their calf muscle activation pattern when provided with implicit biofeedback-driven gaming in an enjoyable manner. Follow-up gait training studies can incorporate this method to assess retention and long-term functional benefits of electromyographic biofeedback-driven gaming.

Towards automated video-based assessment of dystonia in dyskinetic cerebral palsy: A novel approach using markerless motion tracking and machine learning.

Introduction: Video-based clinical rating plays an important role in assessing dystonia and monitoring the effect of treatment in dyskinetic cerebral palsy (CP). However, evaluation by clinicians is time-consuming, and the quality of rating is dependent on experience. The aim of the current study is to provide a proof-of-concept for a machine learning approach to automatically assess scoring of dystonia using 2D stick figures extracted from videos. Model performance was compared to human performance. Methods: A total of 187 video sequences of 34 individuals with dyskinetic CP (8-23 years, all non-ambulatory) were filmed at rest during lying and supported sitting. Videos were scored by three raters according to the Dyskinesia Impairment Scale (DIS) for arm and leg dystonia (normalized scores ranging from 0-1). Coordinates in pixels of the left and right wrist, elbow, shoulder, hip, knee and ankle were extracted using DeepLabCut, an open source toolbox that builds on a pose estimation algorithm. Within a subset, tracking accuracy was assessed for a pretrained human model and for models trained with an increasing number of manually labeled frames. The mean absolute error (MAE) between DeepLabCut's prediction of the position of body points and manual labels was calculated. Subsequently, movement and position features were calculated from extracted body point coordinates. These features were fed into a Random Forest Regressor to train a model to predict the clinical scores. The model performance trained with data from one rater evaluated by MAEs (model-rater) was compared to inter-rater accuracy. Results: A tracking accuracy of 4.5 pixels (approximately 1.5 cm) could be achieved by adding 15-20 manually labeled frames per video. The MAEs for the trained models ranged from 0.21 ± 0.15 for arm dystonia to 0.14 ± 0.10 for leg dystonia (normalized DIS scores). The inter-rater MAEs were 0.21 ± 0.22 and 0.16 ± 0.20, respectively. Conclusion: This proof-of-concept study shows the potential of using stick figures extracted from common videos in a machine learning approach to automatically assess dystonia. Sufficient tracking accuracy can be reached by manually adding labels within 15-20 frames per video. With a relatively small data set, it is possible to train a model that can automatically assess dystonia with a performance comparable to human scoring.

The influence of wearing an ultrasound device on gait in children with cerebral palsy and typically developing children.

BACKGROUND: Ultrasonography with motion analysis enables dynamic imaging of medial gastrocnemius (MG) muscles and tendons during gait. This revealed pathological muscle-tendon dynamics in children with spastic cerebral palsy (CP) compared to typically developing (TD) children. However, wearing an ultrasound probe on the lower leg could interfere with gait and bias muscle length changes observed with ultrasound. RESEARCH QUESTION: Does wearing an ultrasound probe on the MG influence gait in children with CP and TD children? METHODS: Eighteen children with spastic CP and 16 age-matched TD children walked at comfortable walking speed on an instrumented treadmill. One baseline gait condition (BASE) and two conditions with an ultrasound probe and custom-made probe holder were measured: on the mid-muscle fascicles (FAS) and on the muscle-tendon junction (MTJ). The effect of condition and group on spatiotemporal parameters, hip, knee and ankle kinematics, ankle moment, ankle power, and modeled MG muscle-tendon unit (MTU) length was assessed using two-way repeated measures ANOVA's. Statistical non-parametric mapping was applied for time-series. Post-hoc paired-samples t-tests were conducted, and the root mean square difference was calculated for significant parts. RESULTS: Children took wider steps during FAS (CP, TD) and MTJ (TD) compared to BASE, and during FAS compared to MTJ (CP). Hip extension was lower (2.7°) during terminal stance for MTJ compared to FAS for TD only. There was less swing knee flexion (FAS 4.9°; MTJ 4.0°) and ankle plantarflexion around toe-off (FAS 3.0°; MTJ 2.4°) for both ultrasound placements, with no group effect. Power absorption during loading response was slightly increased for both ultrasound placements (0.12 W/kg), with no group effect. MTU shortened less in swing for both ultrasound placements (FAS 3.6 mm; MTJ 3.7 mm), with no group effect. SIGNIFICANCE: Wearing an ultrasound probe causes minimal lower-limb gait alterations and MTU length changes that are mostly similar in CP and TD.

Brain structural and functional connectivity and network organization in cerebral palsy: A scoping review.

AIM: To explore altered structural and functional connectivity and network organization in cerebral palsy (CP), by clinical CP subtype (unilateral spastic, bilateral spastic, dyskinetic, and ataxic CP). METHOD: PubMed and Embase databases were systematically searched. Extracted data included clinical characteristics, analyses, outcome measures, and results. RESULTS: Sixty-five studies were included, of which 50 investigated structural connectivity, and 20 investigated functional connectivity using functional magnetic resonance imaging (14 studies) or electroencephalography (six studies). Five of the 50 studies of structural connectivity and one of 14 of functional connectivity investigated whole-brain network organization. Most studies included patients with unilateral spastic CP; none included ataxic CP. INTERPRETATION: Differences in structural and functional connectivity were observed between investigated clinical CP subtypes and typically developing individuals on a wide variety of measures, including efferent, afferent, interhemispheric, and intrahemispheric connections. Directions for future research include extending knowledge in underrepresented CP subtypes and methodologies, evaluating the prognostic potential of specific connectivity and network measures in neonates, and understanding therapeutic effects on brain connectivity.