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Abstract Classifying gait patterns into homogeneous groups could enhance communication among healthcare providers, clinical decision making and clinical trial designs in boys with Duchenne muscular dystrophy (DMD). Sutherland’s classification has been developed 40 years ago. Ever since, the state-of-the-art medical care has improved and boys with DMD are now longer ambulatory. Therefore, the gait classification requires an update. The overall aim was to develop an up-to-date, valid DMD gait classification. A total of 137 three-dimensional gait analysis sessions were collected in 30 boys with DMD, aged 4.6–17 years. Three classes were distinguished, which only partly aligned with increasing severity of gait deviations. Apart from the mildly affected pattern, two more severely affected gait patterns were found, namely the tiptoeing pattern and the flexion pattern with distinct anterior pelvic tilt and posterior trunk leaning, which showed most severe deviations at the ankle or at the proximal segments/joints, respectively. The agreement between Sutherland’s and the current classification was low, suggesting that gait pathology with the current state-of-the-art medical care has changed. However, overlap between classes, especially between the two more affected classes, highlights the complexity of the continuous gait changes. Therefore, caution is required when classifying individual boys with DMD into classes.

Joint hyper-resistance is a common symptom in neurological disorders. It has both neural and non-neural origins, but it has been challenging to distinguish different origins based on clinical tests alone. Combining instrumented tests with parameter identification based on a neuromechanical model may allow us to dissociate the different origins of joint hyper-resistance in individual patients. However, this requires that the model captures the underlying mechanisms. Here, we propose a neuromechanical model that, in contrast to previously proposed models, accounts for muscle short-range stiffness (SRS) and its interaction with muscle tone and reflex activity. We collected knee angle trajectories during the pendulum test in 15 children with cerebral palsy (CP) and 5 typically developing children. We did the test in two conditions – hold and pre-movement – that have been shown to alter knee movement. We modeled the lower leg as an inverted pendulum actuated by two antagonistic Hill-type muscles extended with SRS. Reflex activity was modeled as delayed, linear feedback from muscle force. We estimated neural and non-neural parameters by optimizing the fit between simulated and measured knee angle trajectories during the hold condition. The model could fit a wide range of knee angle trajectories in the hold condition. The model with personalized parameters predicted the effect of pre-movement demonstrating that the model captured the underlying mechanism and subject-specific deficits. Our model may help with the identification of neural and non-neural origins of joint hyper-resistance and thereby opens perspectives for improved diagnosis and treatment selection in children with spastic CP, but such applications require further studies to establish the method’s reliability.

Ankle–foot orthoses (AFOs) are commonly prescribed to children with cerebral palsy (CP). The conventional AFO successfully controls the first and second ankle rocker, but it fails to correct the third ankle rocker, which negatively effects push-off power. The current study evaluated a new powered AFO (PAFO) design, developed to address the shortcomings of the conventional AFO. Eight children with spastic CP (12.4 ± 3.4 years; GMFCS I-III; 4/4-♂/♀; 3/5-bi/unilateral) were included. Sagittal kinematic and kinetic data were collected from 20 steps during barefoot walking, with conventional AFOs and PAFOs. In the PAFO-condition, an actuation unit was attached to a hinged AFO and through push–pull cables to a backpack that was carried by the child and provided patient-specific assistance-as-needed. SnPM-analysis indicated gait cycle sections that differed significantly between conditions. For the total group, differences between the three conditions were found in ankle kinematics (49.6–66.1%, p = 0.006; 88.0–100%, p = 0.011) and angular velocity (0.0–6.0%, p = 0.001; 45.1–51.1%, p = 0.006; 62.2–73.0%, p = 0.001; 81.2–93.0%, p = 0.001). Individual SnPM-analysis revealed a greater number of significant gait cycle sections for kinematics and kinetics of the ankle, knee, and hip. These individual results were heterogeneous and specific per gait pattern. In conclusion, the new PAFO improved the ankle range-of-motion, angular velocity, and power during push-off in comparison to the conventional AFO.

Balance impairments are common in cerebral palsy. When balance is perturbed by backward support surface translations, children with cerebral palsy have increased co-activation of the plantar flexors and tibialis anterior muscle as compared to typically developing children. However, it is unclear whether increased muscle co-activation is a compensation strategy to improve balance control or is a consequence of reduced reciprocal inhibition. During translational perturbations, increased joint stiffness due to co-activation might aid balance control by resisting movement of the body with respect to the feet. In contrast, during rotational perturbations, increased joint stiffness will hinder balance control as it couples body to platform rotation. Therefore, we expect increased muscle co-activation in response to rotational perturbations if co-activation is caused by reduced reciprocal inhibition but not if it is merely a compensation strategy. We perturbed standing balance by combined backward translational and toe-up rotational perturbations in 20 children with cerebral palsy and 20 typically developing children. Perturbations induced forward followed by backward movement of the center of mass. We evaluated reactive muscle activity and the relation between center of mass movement and reactive muscle activity using a linear feedback model based on center of mass kinematics. In typically developing children, perturbations induced plantar flexor balance correcting muscle activity followed by tibialis anterior balance correcting muscle activity, which was driven by center of mass movement. In children with cerebral palsy, the switch from plantar flexor to tibialis anterior activity was less pronounced than in typically developing children due to increased muscle co-activation of the plantar flexors and tibialis anterior throughout the response. Our results thus suggest that a reduction in reciprocal inhibition causes muscle co-activation in reactive standing balance in children with cerebral palsy.

Introduction: Histological data on muscle fiber size and proportion in (very) young typically developing (TD) children is not well documented and data on capillarization and satellite cell content are also lacking.Aims: This study investigated the microscopic properties of the medial gastrocnemius muscle in growing TD children, grouped according to age and gender to provide normal reference values in healthy children.Methods: Microbiopsies of the medial gastrocnemius (MG) muscle were collected in 46 TD boys and girls aged 2–10 years subdivided into 4 age groups (2–4, 4–6, 6–8 and 8–10 years). Sections were immunostained to assess fiber type cross-sectional area (fCSA) and proportion, the number of satellite cells (SC), capillary to fiber ratio (C/F), capillary density for type I and II fiber (CFD), capillary domain, capillary-to-fiber perimeter exchange index (CFPE) and heterogeneity index. fCSA was normalized to fibula length2 and the coefficient of variation (CV) was calculated to reflect fCSA intrasubject variability.Results: Absolute fCSA of all fibers increased with age (r = 0.72, p < 0.001) but more in boys (+112%, p < 0.05) than in girls (+48%, p > 0.05) Normalized fCSA, CV and fiber proportion did not differ between age groups and gender. C/F was strongly correlated with age in boys (r = 0.83, p < 0.001), and to a lesser extent in girls (r = 0.37, p = 0.115), while other capillary parameters as well as the number of SC remained stable with increasing age in boys and girls.Discussion: This study provides reference values of histological measures in MG according to age in normally growing boys and girls. These data may be used as a reference to determine disease impact and efficacy of therapeutic approach on the muscle.

Different types of feedback are used during gait training in children with cerebral palsy (CP), including verbal (VB) and virtual reality (VR) feedback. Previous studies on VR feedback showed positive effects on the targeted gait parameter. However, both positive and negative side effects on other parameters were seen as well. The literature on the effect of VB feedback is lacking and, to our knowledge, both feedback methods have not yet been compared. In this monocentric study with a single-session intervention protocol, children with CP completed a training session on the Gait Real-Time Analysis Interactive Lab (GRAIL) and received both VB and VR feedback on hip extension, in randomized order. Outcome parameters were continuous gait curves of sagittal kinematics and hip kinetics, specific features of hip angle and moment, sagittal gait variable scores and gait profile scores. Improvement of the targeted gait parameter was seen both after VB and VR feedback, with a small advantage for VR over VB feedback. Furthermore, positive side effects on knee and ankle sagittal kinematics were seen. However, the overall gait profile score did not improve, most likely due to negative compensatory strategies. In conclusion, children with CP can adapt gait in response to both VB and VR feedback, with VR feedback producing a slightly better effect. Due to secondary effects on parameters other than the targeted parameter, the overall gait did not improve.

Botulinum toxin-A (BoNT-A) injection is known to exert beneficial effects on muscle tone, joint mobility and gait in children with cerebral palsy (CP). However, recent animal and human studies have raised the concern that BoNT-A might be harmful to muscle integrity. In CP-children, the impact of BoNT-A on muscle structure has been poorly studied, and inconsistent results have been reported. This study was aimed at determining the time course effect of a single BoNT-A administration on medial gastrocnemius (MG) morphology in CP-children. MG microbiopsies from 12 ambulant and BoNT-A-naïve CP-children (age, 3.4 (2.3) years, ranging from 2.5 to 7.8 years; seven boys and five girls; GMFCS I = 5, II = 4 and III = 3) were collected before and 3 and 6 months after BoNT-A treatment to analyze the fiber cross-sectional area (fCSA) and proportion; capillarization; and satellite cell (SC) content. Compared with the baseline, the fCSA decreased at 3 months (−14%, NS) and increased at 6 months (+13%, NS). Fiber size variability was significantly higher at 3 months (type I: +56%, p = 0.032; type IIa: +37%, p = 0.032) and 6 months (type I: +69%, p = 0.04; type IIa: +121%, p = 0.032) compared with the baseline. The higher type I proportion seen at 3 months was still present and more pronounced at 6 months (type I: +17%, p = 0.04; type IIx: −65%, p = 0.032). The capillary fiber density was reduced at 3 months (type I: −43%, NS; type II: −44%, p = 0.0320) but normalized at 6 months. There was a non-significant increase in SC/100 fibers at 3 months (+75%, NS) and 6 months (+40%, NS) compared with the baseline. These preliminary data suggest that BoNT-A induced alterations in the MG of children with CP, which were still present 6 months after BoNT-A injection but with signs of muscle recovery.

Abstract Background The analysis of in vitro cultures of human adult muscle stem cells obtained from biopsies delineates the potential of skeletal muscles and may help to understand altered muscle morphology in patients. In these analyses, the fusion index is a commonly used quantitative metric to assess the myogenic potency of the muscle stem cells. Since the fusion index only partly describes myogenic potency, we developed the Myotube Analyzer tool, which combines the definition of the fusion index with extra features of myonuclei and myotubes obtained from satellite cell cultures. Results The software contains image adjustment and mask editing functions for preprocessing and semi-automatic segmentation, while other functions can be used to determine the features of nuclei and myotubes. The fusion index and a set of five novel parameters were tested for reliability and validity in a comparison between satellite cell cultures from children with cerebral palsy and typically developing children. These novel parameters quantified extra nucleus and myotube properties and can be used to describe nucleus clustering and myotube shape. Two analyzers who were trained in cell culture defined all parameters using the Myotube Analyzer app. Out of the six parameters, five had good reliability reflected by good intra-class correlation coefficients (> 0.75). Children with cerebral palsy were significantly different from the typically developing children (p

Cerebral palsy (CP) is a heterogeneous group of motor disorders attributed to a non-progressive lesion in the developing brain. Knowledge on skeletal muscle properties is important to understand the impact of CP and treatment but data at the microscopic levels are limited and inconsistent. Currently, muscle biopsies are collected during surgery and are restricted to CP eligible for such treatment or they may refer to another muscle or older children in typically developing (TD) biopsies. A minimally invasive technique to collect (repeated) muscle biopsies in young CP and TD children is needed to provide insights into the early muscle microscopic alterations and their evolution in CP. This paper describes the protocol used to 1) collect microbiopsies of the medial gastrocnemius (MG) and semitendinosus (ST) in CP children and age-matched TD children, 2) handle the biopsies for histology, 3) stain the biopsies to address muscle structure (Hematoxylin & Eosin), fiber size and proportion (myosin heavy chain), counting of the satellite cells (Pax7) and capillaries (CD31). Technique feasibility and safety as well as staining feasibility and measure accuracy were evaluated. Two microbiopsies per muscle were collected in 56 CP (5.8±1.1 yr) and 32 TD (6±1.1 yr) children using ultrasound-guided percutaneous microbiopsy technique. The biopsy procedure was safe (absence of complications) and well tolerated (Score pain using Wong-Baker faces). Cross-sectionally orientated fibers were found in 86% (CP) and 92% (TD) of the biopsies with 60% (CP) and 85% (TD) containing more than 150 fibers. Fiber staining was successful in all MG biopsies but failed in 30% (CP) and 16% (TD) of the ST biopsies. Satellite cell staining was successful in 89% (CP) and 85% (TD) for MG and in 70% (CP) and 90% (TD) for ST biopsies, while capillary staining was successful in 88% (CP) and 100% (TD) of the MG and in 86% (CP) and 90% (TD) for the ST biopsies. Intraclass coefficient correlation showed reliable and reproducible measures of all outcomes. This study shows that the percutaneous microbiopsy technique is a safe and feasible tool to collect (repeated) muscle biopsies in young CP and TD children for histological analysis and it provides sufficient muscle tissue of good quality for reliable quantification.

Background: Studies aiming to objectively quantify movement disorders during upper limb tasks using wearable sensors have recently increased, but there is a wide variety in described measurement and analyzing methods, hampering standardization of methods in research and clinics. Therefore, the primary objective of this review was to provide an overview of sensor set-up and type, included tasks, sensor features and methods used to quantify movement disorders during upper limb tasks in multiple pathological populations. The secondary objective was to identify the most sensitive sensor features for the detection and quantification of movement disorders on the one hand and to describe the clinical application of the proposed methods on the other hand.Methods: A literature search using Scopus, Web of Science, and PubMed was performed. Articles needed to meet following criteria: 1) participants were adults/children with a neurological disease, 2) (at least) one sensor was placed on the upper limb for evaluation of movement disorders during upper limb tasks, 3) comparisons between: groups with/without movement disorders, sensor features before/after intervention, or sensor features with a clinical scale for assessment of the movement disorder. 4) Outcome measures included sensor features from acceleration/angular velocity signals.Results: A total of 101 articles were included, of which 56 researched Parkinson’s Disease. Wrist(s), hand(s) and index finger(s) were the most popular sensor locations. Most frequent tasks were: finger tapping, wrist pro/supination, keeping the arms extended in front of the body and finger-to-nose. Most frequently calculated sensor features were mean, standard deviation, root-mean-square, ranges, skewness, kurtosis/entropy of acceleration and/or angular velocity, in combination with dominant frequencies/power of acceleration signals. Examples of clinical applications were automatization of a clinical scale or discrimination between a patient/control group or different patient groups.Conclusion: Current overview can support clinicians and researchers in selecting the most sensitive pathology-dependent sensor features and methodologies for detection and quantification of upper limb movement disorders and objective evaluations of treatment effects. Insights from Parkinson’s Disease studies can accelerate the development of wearable sensors protocols in the remaining pathologies, provided that there is sufficient attention for the standardisation of protocols, tasks, feasibility and data analysis methods.

The aim of this study was to determine the clinimetric properties, i.e., reliability, validity and responsiveness of an instrumented strength assessment in typically developing (TD) children and children with cerebral palsy (CP) and Duchenne muscular dystrophy (DMD). Force (N), torque (Nm) and normalized torque (Nm/kg) were defined for maximal voluntary isometric contractions (MVICs) of the lower limb muscles using a pre-established protocol. Intraclass correlation coefficient (ICC), standard error of measurement (SEM) and minimal detectable change (MDC) of TD children (n = 14), children with CP (n = 11) and DMD (n = 11) were used to evaluate intra-rater reliability for the three cohorts and the inter-rater intersession as well as inter-rater intrasession reliability for TD children. Construct validity was assessed by comparing MVICs in TD children (n = 28) to children with CP (n = 26) and to children with DMD (n = 30), using the Kruskal Wallis and post-hoc Mann-Whitney U tests. Responsiveness was investigated by assessing changes in MVICs following a strength intervention in CP (n = 26) and a 1 and 2 year follow-up study in DMD (n = 13 and n = 6, respectively), using the Wilcoxon Signed-Rank test. The overall intra-rater reliability, was classified as good to excellent for 65.1%, moderate for 27.0% and poor for 7.9% of the measures (47.6%, 76.2%, and 66.7% good-excellent; 28.6%, 23.8%, and 33.7% moderate; 23.8%, 0%, and 0% poor in TD, CP, and DMD, respectively), while ICC values for TD children were slightly lower for inter-rater intrasession reliability (38.1% good-excellent, 33.3% moderate and 26.6% poor) and for inter-rater intersession reliability (47.6% good-excellent, 23.8% moderate and 28.6% poor). Children with CP and DMD were significantly weaker than TD children (p < 0.001) and the majority of these strength differences exceeded the MDC. Children with CP significantly improved strength after training, with changes that exceeded the SEMs, whereas only limited strength decreases over time were observed in the DMD cohort. In conclusion, the investigated instrumented strength assessment was sufficiently reliable to confirm known-group validity for both cohorts and could detect the responsiveness of children with CP after a strength intervention. However, more research is necessary to determine the responsiveness of this assessment in children with DMD regarding their natural decline.

Children with spastic cerebral palsy often present with muscle weakness, resulting from neural impairments and muscular alterations. While progressive resistance training (PRT) improves muscle weakness, the effects on muscle morphology remain inconclusive. This investigation evaluated the effects of a PRT program on lower limb muscle strength, morphology and gross motor function. Forty-nine children with spastic cerebral palsy were randomized by minimization. The intervention group (nparticipants = 26, age: 8.3 ± 2.0 years, Gross Motor Function Classification System [GMFCS] level I/II/III: 17/5/4, nlegs = 41) received a 12-week PRT program, consisting of 3-4 sessions per week, with exercises performed in 3 sets of 10 repetitions, aiming at 60%–80% of the 1-repetition maximum. Training sessions were performed under supervision with the physiotherapist and at home. The control group (nparticipants = 22, age: 8.5 ± 2.1 year, GMFCS level I/II/III: 14/5/3, nlegs = 36) continued usual care including regular physiotherapy and use of orthotics. We assessed pre- and post-training knee extension, knee flexion and plantar flexion isometric strength, rectus femoris, semitendinosus and medial gastrocnemius muscle morphology, as well as functional strength, gross motor function and walking capacity. Data processing was performed blinded. Linear mixed models were applied to evaluate the difference in evolution over time between the control and intervention group (interaction-effect) and within each group (time-effect). The α-level was set at p = 0.01. Knee flexion strength and unilateral heel raises showed a significant interaction-effect (p ≤ 0.008), with improvements in the intervention group (p ≤ 0.001). Moreover, significant time-effects were seen for knee extension and plantar flexion isometric strength, rectus femoris and medial gastrocnemius MV, sit-to-stand and lateral step-up in the intervention group (p ≤ 0.004). Echo-intensity, muscle lengths and gross motor function showed limited to no changes. PRT improved strength and MV in the intervention group, whereby strength parameters significantly or close to significantly differed from the control group. Although, relative improvements in strength were larger than improvements in MV, important effects were seen on the maintenance of muscle size relative to skeletal growth. In conclusion, this study proved the effectiveness of a home-based, physiotherapy supervised, PRT program to improve isometric and functional muscle strength in children with SCP without negative effects on muscle properties or any serious adverse events.Clinical Trial Registration: ClinicalTrials.gov, identifier NCT03863197.

Botulinum neurotoxin type-A (BoNT) injections are commonly used as spasticity treatment in cerebral palsy (CP). Despite improved clinical outcomes, concerns regarding harmful effects on muscle morphology have been raised, and the BoNT effect on muscle stem cells remains not well defined. This study aims at clarifying the impact of BoNT on growing muscles (1) by analyzing the in vitro effect of BoNT on satellite cell (SC)-derived myoblasts and fibroblasts obtained from medial gastrocnemius microbiopsies collected in young BoNT-naïve children (t0) compared to age ranged typically developing children; (2) by following the effect of in vivo BoNT administration on these cells obtained from the same children with CP at 3 (t1) and 6 (t2) months post BoNT; (3) by determining the direct effect of a single and repeated in vitro BoNT treatment on neuromuscular junctions (NMJs) differentiated from hiPSCs. In vitro BoNT did not affect myogenic differentiation or collagen production. The fusion index significantly decreased in CP at t2 compared to t0. In NMJ cocultures, BoNT treatment caused axonal swelling and fragmentation. Repeated treatments impaired the autophagic–lysosomal system. Further studies are warranted to understand the long-term and collateral effects of BoNT in the muscles of children with CP.

Prolonging ambulation is an important treatment goal in children with Duchenne muscular dystrophy (DMD). Three-dimensional gait analysis (3DGA) could provide sensitive parameters to study the efficacy of clinical trials aiming to preserve ambulation. However, quantitative descriptions of the natural history of gait features in DMD are first required. The overall goal was to provide a full delineation of the progressive gait pathology in children with DMD, covering the entire period of ambulation, by performing a so-called mixed cross-sectional longitudinal study. Firstly, to make our results comparable with previous literature, we aimed to cross-sectionally compare 31 predefined gait features between children with DMD and a typically developing (TD) database (1). Secondly, we aimed to explore the longitudinal changes in the 31 predefined gait features in growing boys with DMD using follow-up 3DGA sessions (2). 3DGA-sessions (n = 124) at self-selected speed were collected in 27 boys with DMD (baseline age: 4.6–15 years). They were repeatedly measured over a varying follow-up period (range: 6 months–5 years). The TD group consisted of 27 children (age: 5.4–15.6 years). Per measurement session, the spatiotemporal parameters, and the kinematic and kinetic waveforms were averaged over the selected gait cycles. From the averaged waveforms, discrete gait features (e.g., maxima and minima) were extracted. Mann-Whitney U tests were performed to cross-sectionally analyze the differences between DMD at baseline and TD (1). Linear mixed effect models were performed to assess the changes in gait features in the same group of children with DMD from both a longitudinal (i.e., increasing time) as well as a cross-sectional perspective (i.e., increasing baseline age) (2). At baseline, the boys with DMD differed from the TD children in 17 gait features. Additionally, 21 gait features evolved longitudinally when following-up the same boys with DMD and 25 gait features presented a significant cross-sectional baseline age-effect. The current study quantitatively described the longitudinal alterations in gait features in boys with DMD, thereby providing detailed insight into how DMD gait deteriorates. Additionally, our results highlight that gait features extracted from 3DGA are promising outcome measures for future clinical trials to quantify the efficacy of novel therapeutic strategies.

BackgroundThis study aimed to analyze the reliability of concentric isokinetic strength assessments (knee and hip) using the Biodex System 4 in healthy children and assess the association with functional strength tests (sit-to-stand [STS], lateral-step-up [LSU]).Methods19 children (6–12 years) were included. Knee and hip flexion and extension, and hip abduction and adduction were tested at 60 and 90°/s.ResultsRelative and absolute reliability at 60°/s tended to show better results compared to those at 90°/s. Intra class correlations (ICCs) of knee flexion and extension at 60°/s were good (0.79–0.89). For hip flexion, extension, abduction and adduction at 60°/s ICCs were moderate to good (0.53–0.83). The smallest detectable change (SDC) values (expressed in %) were highly variable. The SDC% for knee flexion and extension and hip abduction at 60°/s were around 50%. Positive associations were found between hip extension and abduction isokinetic strength and the STS test.ConclusionConcentric isokinetic strength assessments in healthy children using the Biodex System 4 were found reliable for knee flexion and extension and hip abduction. Limited associations were found between concentric isokinetic strength tests and functional strength tests.

Dyskinetic cerebral palsy (DCP) is characterised by involuntary movements, and the movement patterns of children with DCP have not been extensively studied during upper limb tasks. The aim of this study is to evaluate psychometric properties of upper limb kinematics in participants with DCP and typically developing (TD) participants. In current repeatability and validity study, forty individuals with typical development (n = 20) and DCP (n = 20) performed a reach forward/sideways and a reach and grasp task during motion analysis on two occasions. Joint angles at point of task achievement (PTA) and spatio-temporal parameters were evaluated within-and between-sessions using intra-class correlation coefficients (ICC) and standard error of measurement (SEM). Independent t-tests/Mann-Whitney-U tests were used to compare parameters between groups. Within-session ICC values ranged from 0.45 to 1.0 for all parameters for both groups. Within-session SEM values ranged from 1.1° to 11.7° for TD participants and from 1.9° to 13.0° for participants with DCP. Eight within-session repetitions resulted in the smallest change in ICC and SEM values for both groups. Within-session variability was higher for participants with DCP in comparison with the TD group for the majority of the joint angles and spatio-temporal parameters. Intrinsic variability over time was small for all angles and spatio-temporal parameters, whereas extrinsic variability was higher for elbow and scapula angles. Between-group differences revealed lower shoulder adduction and higher elbow flexion, pronation and wrist flexion, as well as higher trajectory deviation and a lower maximal velocity for participants with DCP. This is the first study to assess the psychometric properties of upper limb kinematics in children and adolescents with DCP, showing that children with DCP show higher variability during task execution, requiring a minimum of eight repetitions. However, their variable movement pattern can be reliably captured within-and between-sessions, confirming the potential of three-dimensional motion analysis for assessment of rehabilitation interventions in DCP.

Background—Movement patterns in dyskinetic cerebral palsy (DCP) are characterized by abnormal postures and involuntary movements. Current evaluation tools in DCP are subjective and time-consuming. Sensors could yield objective information on pathological patterns in DCP, but their reliability has not yet been evaluated. The objectives of this study were to evaluate (i) reliability and (ii) discriminative ability of sensor parameters. Methods—Inertial measurement units were placed on the arm, forearm, and hand of individuals with and without DCP while performing reach-forward, reach-and-grasp-vertical, and reach-sideways tasks. Intra-class correlation coefficients (ICC) were calculated for reliability, and Mann–Whitney U-tests for between-group differences. Results—Twenty-two extremities of individuals with DCP (mean age 16.7 y) and twenty individuals without DCP (mean age 17.2 y) were evaluated. ICC values for all sensor parameters except jerk and sample entropy ranged from 0.50 to 0.98 during reach forwards/sideways and from 0.40 to 0.95 during reach-and-grasp vertical. Jerk and maximal acceleration/angular velocity were significantly higher for the DCP group in comparison with peers. Conclusions—This study was the first to assess the reliability of sensor parameters in individuals with DCP, reporting high between- and within-session reliability for the majority of the sensor parameters. These findings suggest that pathological movements of individuals with DCP can be reliably captured using a selection of sensor parameters.

Hereditary spastic paraplegia (HSP) is a heterogeneous group of genetic neurodegenerative disorders, characterized by progressive lower limb spasticity and weakness resulting from retrograde axonal degeneration of motor neurons (MNs). Here, we generated in vitro human neuromuscular junctions (NMJs) from five HSP patient-specific induced pluripotent stem cell (hiPSC) lines, by means of microfluidic strategy, to model disease-relevant neuropathologic processes. The strength of our NMJ model lies in the generation of lower MNs and myotubes from autologous hiPSC origin, maintaining the genetic background of the HSP patient donors in both cell types and in the cellular organization due to the microfluidic devices. Three patients characterized by a mutation in the SPG3a gene, encoding the ATLASTIN GTPase 1 protein, and two patients with a mutation in the SPG4 gene, encoding the SPASTIN protein, were included in this study. Differentiation of the HSP-derived lines gave rise to lower MNs that could recapitulate pathological hallmarks, such as axonal swellings with accumulation of Acetyl-α-TUBULIN and reduction of SPASTIN levels. Furthermore, NMJs from HSP-derived lines were lower in number and in contact point complexity, denoting an impaired NMJ profile, also confirmed by some alterations in genes encoding for proteins associated with microtubules and responsible for axonal transport. Considering the complexity of HSP, these patient-derived neuronal and skeletal muscle cell co-cultures offer unique tools to study the pathologic mechanisms and explore novel treatment options for rescuing axonal defects and diverse cellular processes, including membrane trafficking, intracellular motility and protein degradation in HSP.

Muscle weakness is a common clinical symptom in children with spastic cerebral palsy (SCP). It is caused by impaired neural ability and altered intrinsic capacity of the muscles. To define the contribution of decreased muscle size to muscle weakness, two cohorts were recruited in this cross-sectional investigation: 53 children with SCP [median age, 8.2 (IQR, 4.1) years, 19/34 uni/bilateral] and 31 children with a typical development (TD) [median age, 9.7 (IQR, 2.9) years]. Muscle volume (MV) and muscle belly length for m. rectus femoris, semitendinosus, gastrocnemius medialis, and tibialis anterior were defined from three-dimensional freehand ultrasound acquisitions. A fixed dynamometer was used to assess maximal voluntary isometric contractions for knee extension, knee flexion, plantar flexion, and dorsiflexion from which maximal joint torque (MJT) was calculated. Selective motor control (SMC) was assessed on a 5-point scale for the children with SCP. First, the anthropometrics, strength, and muscle size parameters were compared between the cohorts. Significant differences for all muscle size and strength parameters were found (p ≤ 0.003), except for joint torque per MV for the plantar flexors. Secondly, the associations of anthropometrics, muscle size, gross motor function classification system (GMFCS) level, and SMC with MJT were investigated using univariate and stepwise multiple linear regressions. The associations of MJT with growth-related parameters like age, weight, and height appeared strongest in the TD cohort, whereas for the SCP cohort, these associations were accompanied by associations with SMC and GMFCS. The stepwise regression models resulted in ranges of explained variance in MJT from 29.3 to 66.3% in the TD cohort and from 16.8 to 60.1% in the SCP cohort. Finally, the MJT deficit observed in the SCP cohort was further investigated using the TD regression equations to estimate norm MJT based on height and potential MJT based on MV. From the total MJT deficit, 22.6–57.3% could be explained by deficits in MV. This investigation confirmed the disproportional decrease in muscle size and muscle strength around the knee and ankle joint in children with SCP, but also highlighted the large variability in the contribution of muscle size to muscle weakness.

Abstract Background Children with cerebral palsy (CP) have altered synergies compared to typically-developing peers, reflecting different neuromuscular control strategies used to move. While these children receive a variety of treatments to improve gait, whether synergies change after treatment, or are associated with treatment outcomes, remains unknown. Methods We evaluated synergies for 147 children with CP before and after three common treatments: botulinum toxin type-A injection (n = 52), selective dorsal rhizotomy (n = 38), and multi-level orthopaedic surgery (n = 57). Changes in synergy complexity were measured by the number of synergies required to explain > 90% of the total variance in electromyography data and total variance accounted for by one synergy. Synergy weights and activations before and after treatment were compared using the cosine similarity relative to average synergies of 31 typically-developing (TD) peers. Results There were minimal changes in synergies after treatment despite changes in walking patterns. Number of synergies did not change significantly for any treatment group. Total variance accounted for by one synergy increased (i.e., moved further from TD peers) after botulinum toxin type-A injection (1.3%) and selective dorsal rhizotomy (1.9%), but the change was small. Synergy weights did not change for any treatment group (average 0.001 ± 0.10), but synergy activations after selective dorsal rhizotomy did change and were less similar to TD peers (− 0.03 ± 0.07). Only changes in synergy activations were associated with changes in gait kinematics or walking speed after treatment. Children with synergy activations more similar to TD peers after treatment had greater improvements in gait. Conclusions While many of these children received significant surgical procedures and prolonged rehabilitation, the minimal changes in synergies after treatment highlight the challenges in altering neuromuscular control in CP. Development of treatment strategies that directly target impaired control or are optimized to an individual’s unique control may be required to improve walking function.

The intra-subject, the inter-operator, and the inter-laboratory variabilities are the main sources of uncertainties in gait analysis, and their effects have been partially described in the literature for adult populations. This study aimed to extend the repeatability and reproducibility analysis to a pediatric population, accounting for the effects induced by the intra-subject variations, the measurement setup, the marker set configuration, and the involved operators in placing markers and EMG electrodes. We evaluated kinematic, kinetic and EMG outputs collected from gait analyses performed on two healthy children in two laboratories, by two operators, and with two marker placement protocols. The two involved centers previously defined a common acquisition procedure based on their routine pipelines. The similarity of kinematic, kinetic, and EMG curves were evaluated by means of the coefficients of the Linear Fit Method, and the Mean Absolute Variability with and without the offset among curves. The inter-operator variability was found to be the main contribution to the overall reproducibility of kinematic and kinetic gait data. On the contrary, the main contribution to the variability of the EMG signals was the intra-subject repeatability that is due to the physiological stride to stride muscle activation variability.

Children with spastic cerebral palsy (SCP) are often treated with intramuscular Botulinum Neurotoxin type-A (BoNT-A). Recent studies demonstrated BoNT-A-induced muscle atrophy and variable effects on gait pathology. This group-matched controlled study in children with SCP compared changes in muscle morphology 8–10 weeks post-BoNT-A treatment (n = 25, median age 6.4 years, GMFCS level I/II/III (14/9/2)) to morphological changes of an untreated control group (n = 20, median age 7.6 years, GMFCS level I/II/III (14/5/1)). Additionally, the effects on gait and spasticity were assessed in all treated children and a subgroup (n = 14), respectively. BoNT-A treatment was applied following an established integrated approach. Gastrocnemius and semitendinosus volume and echogenicity intensity were assessed by 3D-freehand ultrasound, spasticity was quantified through electromyography during passive muscle stretches at different velocities. Ankle and knee kinematics were evaluated by 3D-gait analysis. Medial gastrocnemius (p = 0.018, −5.2%) and semitendinosus muscle volume (p = 0.030, −16.2%) reduced post-BoNT-A, but not in the untreated control group, while echogenicity intensity did not change. Spasticity reduced and ankle gait kinematics significantly improved, combined with limited effects on knee kinematics. This study demonstrated that BoNT-A reduces spasticity and partly improves pathological gait but reduces muscle volume 8–10 weeks post-injections. Close post-BoNT-A follow-up and well-considered treatment selection is advised before BoNT-A application in SCP.

In neurological diseases, muscles often become hyper-resistant to stretch due to hyperreflexia, an exaggerated stretch reflex response that is considered to primarily depend on the muscle's stretch velocity. However, there is still limited understanding of how different biomechanical triggers applied during clinical tests evoke these reflex responses. We examined the effect of imposing a rotation with increasing velocity vs. increasing acceleration on triceps surae muscle repsonse in children with spastic paresis (SP) and compared the responses to those measured in typically developing (TD) children. A motor-operated ankle manipulator was used to apply different bell-shaped movement profiles, with three levels of maximum velocity (70, 110, and 150°/s) and three levels of maximum acceleration (500, 750, and 1,000°/s2). For each profile and both groups, we evaluated the amount of evoked triceps surae muscle activation. In SP, we evaluated two additional characteristics: the intensity of the response (peak EMG burst) and the time from movement initiation to onset of the EMG burst. As expected, the amount of evoked muscle activation was larger in SP compared to TD (all muscles: p < 0.001) and only sensitive to biomechanical triggers in SP. Further investigation of the responses in SP showed that peak EMG bursts increased in profiles with higher peak velocity (lateral gastrocnemius: p = 0.04), which was emphasized by fair correlations with increased velocity at EMG burst onset (all muscles: r > 0.33–0.36, p ≤ 0.008), but showed no significant effect for acceleration. However, the EMG burst was evoked faster with higher peak acceleration (all muscles p < 0.001) whereas it was delayed in profiles with higher peak velocity (medial gastrocnemius and soleus: p < 0.006). We conclude that while exaggerated response intensity (peak EMG burst) seems linked to stretch velocity, higher accelerations seem to evoke faster responses (time to EMG burst onset) in triceps surae muscles in SP. Understanding and controlling for the distinct effects of different biological triggers, including velocity, acceleration but also length and force of the applied movement, will contribute to the development of more precise clinical measurement tools. This is especially important when aiming to understand the role of hyperreflexia during functional movements where the biomechanical inputs are multiple and changing.

Hereditary spastic paraplegia (HSP) is a neurological, genetic disorder that predominantly presents with lower limb spasticity and muscle weakness. Pediatric pure HSP types with infancy or childhood symptom onset resemble in clinical presentation to children with bilateral spastic cerebral palsy (SCP). Hence, treatment approaches in these patient groups are analogous. Altered muscle characteristics, including reduced medial gastrocnemius (MG) muscle growth and hyperreflexia have been quantified in children with SCP, using 3D-freehand ultrasound (3DfUS) and instrumented assessments of hyperreflexia, respectively. However, these muscle data have not yet been studied in children with HSP. Therefore, we aimed to explore these MG muscle characteristics in HSP and to test the hypothesis that these data differ from those of children with SCP and typically developing (TD) children. A total of 41 children were retrospectively enrolled including (1) nine children with HSP (ages of 9–17 years with gross motor function levels I and II), (2) 17 age-and severity-matched SCP children, and (3) 15 age-matched typically developing children (TD). Clinically, children with HSP showed significantly increased presence and severity of ankle clonus compared with SCP (p = 0.009). Compared with TD, both HSP and SCP had significantly smaller MG muscle volume normalized to body mass (p ≤ 0.001). Hyperreflexia did not significantly differ between the HSP and SCP group. In addition to the observed pathological muscle activity for both the low-velocity and the change in high-velocity and low-velocity stretches in the two groups, children with HSP tended to present higher muscle activity in response to increased stretch velocity compared with those with SCP. This exploratory study is the first to reveal MG muscle volume deficits in children with HSP. Moreover, high-velocity-dependent hyperreflexia and ankle clonus is observed in children with HSP. Instrumented impairment assessments suggested similar altered MG muscle characteristics in pure HSP type with pediatric onset compared to bilateral SCP. This finding needs to be confirmed in larger sample sizes. Hence, the study results might indicate analogous treatment approaches in these two patient groups.

Cerebral palsy (CP), the single largest cause of childhood physical disability, is characterized firstly by a lesion in the immature brain, and secondly by musculoskeletal problems that progress with age. Previous research reported altered muscle properties, such as reduced volume and satellite cell (SC) numbers and hypertrophic extracellular matrix compared to typically developing (TD) children (>10 years). Unfortunately, data on younger CP patients are scarce and studies on SCs and other muscle stem cells in CP are insufficient or lacking. Therefore, it remains difficult to understand the early onset and trajectory of altered muscle properties in growing CP children. Because muscle stem cells are responsible for postnatal growth, repair and remodeling, multiple adult stem cell populations from young CP children could play a role in altered muscle development. To this end, new methods for studying muscle samples of young children, valid to delineate the features and to elucidate the regenerative potential of muscle tissue, are necessary. Using minimal invasive muscle microbiopsy, which was applied in young subjects under general anaesthesia for the first time, we aimed to isolate and characterize muscle stem cell-derived progenitors of TD children and patients with CP. Data of 15 CP patients, 3–9 years old, and 5 aged-matched TD children were reported. The muscle microbiopsy technique was tolerated well in all participants. Through the explant technique, we provided muscle stem cell-derived progenitors from the Medial Gastrocnemius. Via fluorescent activated cell sorting, using surface markers CD56, ALP, and PDGFRa, we obtained SC-derived progenitors, mesoangioblasts and fibro-adipogenic progenitors, respectively. Adipogenic, skeletal, and smooth muscle differentiation assays confirmed the cell identity and ability to give rise to different cell types after appropriate stimuli. Myogenic differentiation in CP SC-derived progenitors showed enhanced fusion index and altered myotube formation based on MYOSIN HEAVY CHAIN expression, as well as disorganization of nuclear spreading, which were not observed in TD myotubes. In conclusion, the microbiopsy technique allows more focused muscle research in young CP patients. Current results show altered differentiation abilities of muscle stem cell-derived progenitors and support the hypothesis of their involvement in CP-altered muscle growth.

The pendulum test assesses quadriceps spasticity by dropping the lower leg of a relaxed patient from the horizontal position and observing limb movement. The first swing excursion (FS) decreases with increasing spasticity severity. Our recent simulation study suggests that the reduced initial swing results from muscle short-range stiffness and its interaction with reflex hyper-excitability. Short-range stiffness emerges from the thixotropic behavior of muscles where fiber stiffness upon stretch increases when the muscle is held isometric. Fiber stiffness might thus be higher during the first swing of the pendulum test than during consecutive swings. In addition, it has recently been suggested that muscle spindle firing reflects fiber force rather than velocity and therefore, reflex activity might depend on fiber stiffness. If this hypothesized mechanism is true, we expect to observe larger first swing excursions and reduced reflex muscle activity when the leg is moved rather than kept isometric before release, especially in patients with increased reflex activity. We performed the pendulum test in 15 children with cerebral palsy (CP) and 15 age-matched typically developing (TD) children in two conditions. In the hold condition, the leg was kept isometric in the extended position before release. In the movement condition, the leg was moved up and down before release to reduce the contribution of short-range stiffness. Knee kinematics and muscle activity were recorded. Moving the leg before release increased first swing excursion (p < 0.001) and this increase was larger in children with CP (21°) than in TD children (8°) (p < 0.005). In addition, pre-movement delayed reflex onset by 87 ms (p < 0.05) and reduced reflex activity as assessed through the area under the curve of rectus femoris electromyography (p < 0.05) in children with CP. The movement history dependence of pendulum kinematics and reflex activity supports our hypothesis that muscle short-range stiffness and its interaction with reflex hyper-excitability contribute to joint hyper-resistance in spastic CP. Our results have implications for standardizing movement history in clinical tests of spasticity and for understanding the role of spasticity in functional movements, where movement history differs from movement history in clinical tests.

The interaction between brain damage and motor function is not yet fully understood in children with spastic cerebral palsy (CP). Therefore, a semi-quantitative MRI (sqMRI) scale was used to explore whether identified brain lesions related to functional abilities and gait pathology in this population. A retrospective cohort of ambulatory children with spastic CP was selected [N = 104; 52 bilateral (bCP) and 52 unilateral (uCP)]. Extent and location-specific scores were defined according to the sqMRI scale guidelines. The gross motor function classification system (GMFCS), the gait profile score (GPS), GPSs per motion plane, gait variable scores (GVS) and multiple-joint (MJ) gait patterns were related to brain lesion scores. In all groups, the global total brain scores correlated to the GPS (total: rs = 0.404, p ≤ 0.001; bCP: rs = 0.335, p ≤ 0.05; uCP: rs = 0.493, p ≤ 0.001). The global total hemispheric scores correlated to the GMFCS (total: rs = 0.392, p ≤ 0.001; bCP: rs = 0.316, p ≤ 0.05; uCP: rs = 0.331, p ≤ 0.05). The laterality scores of the hemispheres in the total group correlated negatively to the GMFCS level (rs = −0.523, p ≤ 0.001) and the GVS-knee sagittal (rs = −0.311, p ≤ 0.01). Lesion location, for the total group demonstrated positive correlations between parietal lobe involvement and the GPS (rs = 0.321, p ≤ 0.001) and between periventricular layer damage and the GMFCS (rs = 0.348, p ≤ 0.001). Involvement of the anterior part of the corpus callosum (CC) was associated with the GVS-hip sagittal in all groups (total: rpb = 0.495, p ≤ 0.001; bCP: rpb = 0.357, p ≤ 0.05; uCP: rpb = 0.641, p ≤ 0.001). The global total hemispheric and laterality of the hemispheres scores differentiated between the minor and both the extension (p ≤ 0.001 and p ≤ 0.001) and flexion (p = 0.016 and p = 0.013, respectively) MJ patterns in the total group. Maximal periventricular involvement and CC intactness were associated with extension patterns (p ≤ 0.05 and p ≤ 0.001, respectively). Current findings demonstrated relationships between brain structure and motor function as well as pathological gait, in this cohort of children with CP. These results might facilitate the timely identification of gait pathology and, ultimately, guide individualized treatment planning of gait impairments in children with CP.

While Botulinum NeuroToxin-A (BoNT-A) injections are frequently used to reduce the effects of hyperactive stretch reflexes in children with cerebral palsy (CP), the effects of this treatment vary strongly. Previous research, combining electromyography (EMG) with motion analysis, defined different patterns of stretch reflex muscle activation in muscles, those that reacted more to a change in velocity (velocity dependent –VD), and those that reacted more to a change in length (length dependent –LD). The aim of this study was to investigate the relation between the types of stretch reflex muscle activation in the semitendinosus with post-BoNT-A outcome as assessed passively and with 3D gait analysis in children with spastic CP. Eighteen children with spastic CP (10 bilaterally involved) between the ages of 12 and 18 years were assessed before and on average, 8 weeks post-treatment. EMG and motion analysis were used to assess the degree and type of muscle activation dependency in the semitendinosus during passive knee extensions performed at different joint angular velocities. Three-dimensional gait analysis was used to assess knee gait kinematics as a measure of functional outcome. Pre-treatment, 9 muscles were classified as VD and 9 as LD, but no differences between the groups were evident in the baseline knee gait kinematics. Post-treatment, stretch reflex muscle activation decreased significantly in both groups but the reduction was more pronounced in those muscles classified pre-treatment as VD (−72% vs. −50%, p = 0.005). In the VD group, these changes were accompanied by greater knee extension at initial contact and during the swing phase of gait. In the LD group, there was significantly increased post-treatment knee hyperextension in late stance. Although results vary between patients, the reduction of stretch reflex muscle activation in the semitendinosus generally translated to an improved functional outcome, as assessed with 3D gait analysis. However, results were less positive for those muscles with pre-treatment length-dependent type of stretch reflex muscle activation. The study demonstrates the relevance of categorizing the type of stretch reflex muscle activation as a possible predictor of treatment response.

Botulinum NeuroToxin-A (BoNT-A) injections to the medial gastrocnemius (MG) and lower-leg casts are commonly combined to treat ankle equinus in children with spastic cerebral palsy (CP). However, the decomposed treatment effects on muscle or tendon structure, stretch reflexes, and joint are unknown. In this study, BoNT-A injections to the MG and casting of the lower legs were applied separately to gain insight into the working mechanisms of the isolated treatments on joint, muscle, and tendon levels. Thirty-one children with spastic CP (GMFCS I-III, age 7.4 ± 2.6 years) received either two weeks of lower-leg casts or MG BoNT-A injections. During full range of motion slow and fast passive ankle rotations, joint resistance and MG stretch reflexes were measured. MG muscle and tendon lengths were assessed at resting and at maximum dorsiflexion ankle angles using 3D-freehand ultrasound. Treatment effects were compared using non-parametric statistics. Associations between the effects on joint and muscle or tendon levels were performed using Spearman correlation coefficients (p < 0.05). Increased joint resistance, measured during slow ankle rotations, was not significantly reduced after either treatment. Additional joint resistance assessed during fast rotations only reduced in the BoNT-A group (−37.6%, p = 0.013, effect size = 0.47), accompanied by a reduction in MG stretch reflexes (−70.7%, p = 0.003, effect size = 0.56). BoNT-A increased the muscle length measured at the resting ankle angle (6.9%, p = 0.013, effect size = 0.53). Joint angles shifted toward greater dorsiflexion after casting (32.4%, p = 0.004, effect size = 0.56), accompanied by increases in tendon length (5.7%, p = 0.039, effect size = 0.57; r = 0.40). No associations between the changes in muscle or tendon lengths and the changes in the stretch reflexes were found. We conclude that intramuscular BoNT-A injections reduced stretch reflexes in the MG accompanied by an increase in resting muscle belly length, whereas casting resulted in increased dorsiflexion without any changes to the muscle length. This supports the need for further investigation on the effect of the combined treatments and the development of treatments that more effectively lengthen the muscle.

Physics-based simulations of walking have the theoretical potential to support clinical decision-making by predicting the functional outcome of treatments in terms of walking performance. Yet before using such simulations in clinical practice, their ability to identify the main treatment targets in specific patients needs to be demonstrated. In this study, we generated predictive simulations of walking with a medical imaging based neuro-musculoskeletal model of a child with cerebral palsy presenting crouch gait. We explored the influence of altered muscle-tendon properties, reduced neuromuscular control complexity, and spasticity on gait dysfunction in terms of joint kinematics, kinetics, muscle activity, and metabolic cost of transport. We modeled altered muscle-tendon properties by personalizing Hill-type muscle-tendon parameters based on data collected during functional movements, simpler neuromuscular control by reducing the number of independent muscle synergies, and spasticity through delayed muscle activity feedback from muscle force and force rate. Our simulations revealed that, in the presence of aberrant musculoskeletal geometries, altered muscle-tendon properties rather than reduced neuromuscular control complexity and spasticity were the primary cause of the crouch gait pattern observed for this child, which is in agreement with the clinical examination. These results suggest that muscle-tendon properties should be the primary target of interventions aiming to restore an upright gait pattern for this child. This suggestion is in line with the gait analysis following muscle-tendon property and bone deformity corrections. Future work should extend this single case analysis to more patients in order to validate the ability of our physics-based simulations to capture the gait patterns of individual patients pre- and post-treatment. Such validation would open the door for identifying targeted treatment strategies with the aim of designing optimized interventions for neuro-musculoskeletal disorders.

When treating children with Cerebral Palsy (CP), computational simulations based on musculoskeletal models have a great potential in assisting the clinical decision-making process towards the most promising treatments. In particular, predictive simulations could be used to predict and compare the functional outcome of a series of candidate interventions. In order to be able to benefit from these predictive simulations however, it is important to know how much information about the post-treatment patient's motor control could be gathered from data available before the intervention. Within this paper, we quantified how much of the muscle activity measured after a treatment could be explained by subject-specific muscle synergies computed from EMG data collected before the intervention. We also investigated whether generic synergies could be used, in case no EMG data is available when running predictive simulations, to reproduce both pre- and post-treatment muscle activity in children with CP. Subject-specific synergies proved to be a good indicator of the patient's post-treatment motor control, explaining on average more than 85% of the post-treatment muscle activity, compared to an average of 94% when applied to the original pre-treatment data. Generic synergies explained 84% of the pre-treatment and 83% of the post-treatment muscle activity on average, but performed relatively well for patients with low selective motor control and poorly in patients with more selectivity. Our results suggest that subject-specific muscle synergies computed from pre-treatment EMG data could be used with confidence to represent the post-treatment motor control of children with CP during walking. In addition, when performing simulations involving patients with a low selective motor control, generic synergies could be a valid alternative.

AimOur aim was to determine if synergy weights and activations are altered in Duchenne muscular dystrophy (DMD) and if these alterations could be linked to muscle weakness.MethodsIn 22 children with DMD and 22 typical developing (TD) children of a similar age, surface electromyography (sEMG) of the gluteus medius, rectus femoris (REF), medial hamstrings, tibialis anterior, and medial gastrocnemius (GAS) were recorded during gait. Muscle weakness was assessed with maximal voluntary isometric contractions (MVIC). Synergies were calculated with non-negative matrix factorization. The number of synergies explaining ≥90% of the variance in the sEMG signals (N90), were extracted and grouped with k-means cluster analysis. We verified differences in weights with a Mann-Whitney U test. Statistical non-parametric mapping (Hotelling's T2 test and two-tailed t-test) was used to assess group differences in synergy activations. We used Spearman's rank correlation coefficients and canonical correlation analysis to assess if weakness was related to modifications in weights and activations, respectively.ResultsFor both groups, average N90 was three. In synergy one, characterized by activity at the beginning of stance, the DMDs showed an increased REF weight (p = 0.001) and decreased GAS weight (p = 0.007). Synergy activations were similar, with only a small difference detected in mid-swing in the combined activations (pConclusionDespite the apparent weakness in DMD, synergy weights and activations were similar between the two groups. Our findings are in line with previous research suggesting non-neural alterations have limited influence on muscle synergies.

Botulinum Neurotoxin type-A (BoNT-A) injections are widely used as first-line spasticity treatment in spastic cerebral palsy (SCP). Despite improved clinical outcomes, concerns regarding harmful effects on muscle morphology have been raised. Yet, the risk of initiating BoNT-A to reduce muscle growth remains unclear. This study investigated medial gastrocnemius (MG) morphological muscle growth in children with SCP (n = 26, median age of 5.2 years (3.5)), assessed by 3D-freehand ultrasound prior to and six months post-BoNT-A injections. Post-BoNT-A MG muscle growth of BoNT-A naive children (n = 11) was compared to (a) muscle growth of children who remained BoNT-A naive after six months (n = 11) and (b) post-BoNT-A follow-up data of children with a history of BoNT-A treatment (n = 15). Six months after initiating BoNT-A injection, 17% decrease in mid-belly cross-sectional area normalized to skeletal growth and 5% increase in echo-intensity were illustrated. These muscle outcomes were only significantly altered when compared with children who remained BoNT-A naive (+4% and −3%, respectively, p < 0.01). Muscle length growth persevered over time. This study showed reduced cross-sectional growth post-BoNT-A treatment suggesting that re-injections should be postponed at least beyond six months. Future research should extend follow-up periods investigating muscle recovery in the long-term and should include microscopic analysis.

Abstract Background Upper limb (UL) deficits in children with unilateral cerebral palsy (uCP) have traditionally been targeted with motor execution treatment models, such as modified Constraint-Induced Movement Therapy (mCIMT). However, new approaches based on a neurophysiological model such as Action-Observation Training (AOT) may provide new opportunities for enhanced motor learning. The aim of this study is to describe a randomised controlled trial (RCT) protocol investigating the effects of an intensive treatment model, combining mCIMT and AOT compared to mCIMT alone on UL function in children with uCP. Additionally, the role of neurological factors as potential biomarkers of treatment response will be analysed. Methods An evaluator-blinded RCT will be conducted in 42 children aged between 6 and 12 years. Before randomization, children will be stratified according to their House Functional Classification Scale, age and type of corticospinal tract wiring. A 2-week day-camp will be set up in which children receive intensive mCIMT therapy for 6 hours a day on 9 out of 11 consecutive days (54 h) including AOT or control condition (15 h). During AOT, these children watch video sequences showing goal-directed actions and subsequently execute the observed actions with the more impaired UL. The control group performs the same actions after watching computer games without human motion. The primary outcome measure will be the Assisting Hand Assessment. Secondary outcomes comprise clinical assessments across body function, activity and participation level of the International Classification of Function, Disability and Health. Furthermore, to quantitatively evaluate UL movement patterns, a three-dimensional motion analysis will be conducted. UL function will be assessed at baseline, immediately before and after intervention and at 6 months follow up. Brain imaging comprising structural and functional connectivity measures as well as Transcranial Magnetic Stimulation (TMS) to evaluate corticospinal tract wiring will be acquired before the intervention. Discussion This paper describes the methodology of an RCT with two main objectives: (1) to evaluate the added value of AOT to mCIMT on UL outcome in children with uCP and (2) to investigate the role of neurological factors as potential biomarkers of treatment response. Trial registration NCT03256357 registered on 21st August 2017 (retrospectively registered).

Neuromusculoskeletal simulation provides a promising platform to inform the design of assistive devices or inform rehabilitation. For these applications, a simulation must be able to accurately represent the person of interest, such as an individual with a neurologic injury. If a simulation fails to predict how an individual recruits and coordinates their muscles during movement, it will have limited utility for informing design or rehabilitation. While inverse dynamic simulations have previously been used to evaluate anticipated responses from interventions, like orthopedic surgery or orthoses, they frequently struggle to accurately estimate muscle activations, even for tasks like walking. The simulated muscle activity often fails to represent experimentally measured muscle activity from electromyographic (EMG) recordings. Research has theorized that the nervous system may simplify the range of possible activations used during dynamic tasks, by constraining activations to weighted groups of muscles, referred to as muscle synergies. Synergies are altered after neurological injury, such as stroke or cerebral palsy (CP), and may provide a method for improving subject-specific models of neuromuscular control. The aim of this study was to test whether constraining simulation to synergies could improve estimated muscle activations compared to EMG data. We evaluated modeled muscle activations during gait for six typically developing (TD) children and six children with CP. Muscle activations were estimated with: (1) static optimization (SO), minimizing muscle activations squared, and (2) synergy SO (SynSO), minimizing synergy activations squared using the weights identified from EMG data for two to five synergies. While SynSO caused changes in estimated activations compared to SO, the correlation to EMG data was not higher in SynSO than SO for either TD or CP groups. The correlations to EMG were higher in CP than TD for both SO (CP: 0.48, TD: 0.36) and SynSO (CP: 0.46, TD: 0.26 for five synergies). Constraining activations to SynSO caused the simulated muscle stress to increase compared to SO for all individuals, causing a 157% increase with two synergies. These results suggest that constraining simulated activations in inverse dynamic simulations to subject-specific synergies alone may not improve estimation of muscle activations during gait for generic musculoskeletal models.

Gait deficits in cerebral palsy (CP) are often treated with a single-event multi-level surgery (SEMLS). Selecting the treatment options (combination of bony and soft tissue corrections) for a specific patient is a complex endeavor and very often treatment outcome is not satisfying. A deterioration in 22.8% of the parameters describing gait performance has been reported and there is need for additional surgery in 11% of the patients. Computational simulations based on musculoskeletal models that allow clinicians to test the effects of different treatment options before surgery have the potential to drastically improve treatment outcome. However, to date, no such simulation and modeling method is available. Two important challenges are the development of methods to include patient-specific neuromechanical impairments into the models and to simulate the effect of different surgical procedures on post-operative gait performance. Therefore, we developed the SimCP framework that allows the evaluation of the effect of different simulated surgeries on gait performance of a specific patient and includes a graphical user interface (GUI) that enables performing virtual surgery on the models. We demonstrated the potential of our framework for two case studies. Models reflecting the patient-specific musculoskeletal geometry and muscle properties are generated based solely on data collected before the treatment. The patient's motor control is described based on muscle synergies derived from pre-operative EMG. The GUI is then used to modify the musculoskeletal properties according to the surgical plan. Since SEMLS does not affect motor control, the same motor control model is used to define gait performance pre- and post-operative. We use the capability gap (CG), i.e., the difference between the joint moments needed to perform healthy walking and the joint moments the personalized model can generate, to quantify gait performance. In both cases, the CG was smaller post- then pre-operative and this was in accordance with the measured change in gait kinematics after treatment.

BACKGROUND:Approximately 50 to 70% of post-stroke subjects present a reduction in the upper limb (UL) function even during the chronic phase. An adjuvant technique widely used in neurorehabilitation is elastic taping applications. However, its efficacy in UL treatment for post-stroke subjects still requires further investigation. OBJECTIVE:To verify the effects of elastic tape (ET) used on the paretic shoulder in upper limb (UL) performance during a drinking task. METHOD:A single-center randomized sham-controlled crossover trial randomized thirteen post-stroke subjects with mild to moderate UL impairment for group allocation to receive first Sham Tape (ST) or first Elastic Tape (ET), with one month of washout. Kinematic measures of a drinking task were taken before and after each intervention (elastic and sham tape), using Three-Dimensional Motion Analysis, and studied using feature analysis and Statistical Parametric Mapping. Outcome measures included spatiotemporal variables, scalar kinematic parameters (starting angles, range of motion-ROM, and endpoint angles) and time-normalized kinematic waveforms of trunk and UL joint angles (scapulothoracic, humerothoracic and elbow). RESULTS:Elastic tape provided common modifications throughout the task (shoulder more towards midline, reduced scapula protraction and trunk flexion) and important alterations at specific time-instants. At the end of the reaching phase, for both groups (ET and ST), the elastic tape increased elbow extension [ET: CI = 12.57 (6.90 to 18.17), p0.05). CONCLUSION:The ET changed UL joint motions and posture during a drinking task in chronic hemiparetic subjects, which defines its role as an adjuvant therapy.

This study aimed to identify the relationships between clinical impairments and gait deviations in children with cerebral palsy (CP). A retrospective convenience sample of 367 children with CP was selected (3-18 years old) and divided in two groups based on clinical symptomatology [unilateral (uCP) / bilateral CP (bCP), (n = 167/200)]. All children underwent a three-dimensional gait analysis and a standardized clinical examination. Gait was inspected on a vector level (all sagittal motions combined), and an individual joint level (pelvis, hip, knee and ankle joint motions). Statistical non-parametric mapping was applied to identify specific parts of the gait cycle displaying relationships between the gait deviations of both groups and the impairment scores of spasticity, weakness, selectivity, and passive range of motion. Impairment scores were summarized in two ways: a) composite impairment scores (e.g. combined spasticity of all assessed muscles acting around the hip, knee and ankle joints) and b) joint specific impairment scores (e.g. spasticity of the muscles acting around the knee joint). Results showed that the vector and most of the individual motions were related to the composite scores. Direct and carry-over relationships were found between certain individual motions and joint impairment scores (around the same or neighboring joints, respectively). All correlations were more prominent for children with bCP compared to uCP, especially regarding the relationships of gait deviations with weakness and reduced selectivity. In conclusion, this study enabled the mapping of relationships between clinical impairments and gait deviations in children with CP, by identifying specific parts of the gait cycle that are related to each of these impairments. These results provide a comprehensive description of these relationships, while simultaneously highlighting the differences between the two CP groups. Integration of these findings could lead to a better understanding of the pathophysiology of gait deviations and, eventually, support individualized treatment planning.

ObjectiveTo test the reliability of the Balance Evaluation Systems Test (BESTest) and Trunk Control Measurement Scale (TCMS) between sessions and raters in the adult spinal deformity (ASD) population.Summary of background dataUp to now evaluation in ASD was mainly based on static radiographic parameters. Recently literature showed that dynamic balance was a better predictor of health-related quality of life than radiographic parameters, stressing the importance of balance assessment. However, to the best of our knowledge, reliability of balance assessment tools has not yet been investigated in the ASD population.MethodsTwenty ASD patients participated in this study. Ten patients were included in the test-retest study, including repeated measurements. Ten patients were measured once, simultaneously but independently by three raters. Each participant performed two balance scales, namely the BESTest and the TCMS. Statistical analysis consisted of intra class correlations (ICC) on scale- and subscale level, and kappa scores on item-level. Cronbach's alpha on total scores, standard errors of measurement (SEM), smallest detectable differences and percentages of agreement were also calculated. Bland-altman plots were created to investigate systematic bias.ResultsICC scores between sessions and raters for TCMS (0.76 and 0.88) and BESTest (0.90 and 0.94) total scores were good to excellent. SEM's between sessions and raters were also low for total scores on TCMS (1.66 and 2.35) and BESTest (2.99 and 2.32). However, on subscale- and item-level reliability decreased and ceiling effects were observed. No systematic bias was observed between sessions and raters.ConclusionBESTest and TCMS showed to be reliable tools to measure balance in ASD on scale-level. However, on subscale- and item-level reliability decreased and ceiling effects were observed. Therefore, the question arises if there is need for an ASD-specific balance scale.

Stretch reflex hyperactivity in the gastrocnemius of children with spastic cerebral palsy (CP) is commonly evaluated by passively rotating the ankle joint into dorsiflexion at different velocities, such as applied in conventional clinical spasticity assessments. However, surface electromyography (sEMG) collected from the medial gastrocnemius (MG) during such examination reveals unexplained heterogeneity in muscle activation between patients. Recent literature also highlights altered muscle tensile behavior in children with spastic CP. We aimed to document MG muscle and tendon lengthening during passive ankle motion at slow and fast velocity and explore its interdependence with the elicited hyperactive stretch reflex. The ankle of 15 children with CP (11 ± 3 years, GMFCS 9I 6II, 8 bilateral, 7 unilateral) and 16 typically developing children (TDC) was passively rotated over its full range of motion at slow and fast velocity. Ultrasound, synchronized with motion-analysis, was used to track the movement of the MG muscle-tendon junction and extract the relative lengthening of muscle and tendon during joint rotation. Simultaneously, MG sEMG was measured. Outcome parameters included the angular and muscle lengthening velocities 30 ms before EMG onset and the gain in root mean square EMG during stretch, as a measure of stretch reflex activity. Compared to slow rotation, the muscle lengthened less and stretch reflex activity was higher during fast rotation. These velocity-induced changes were more marked in CP compared to TDC. In the CP group, muscle-lengthening velocity had higher correlation coefficients with stretch reflex hyperactivity than joint angular velocity. Muscles with greater relative muscle lengthening during slow rotation had earlier and stronger stretch reflexes during fast rotation. These initial results suggest that ankle angular velocity is not representative of MG muscle lengthening velocity and is less related to stretch reflex hyperactivity than MG muscle lengthening. In addition, muscles that lengthened more during slow joint rotation were more likely to show a velocity-dependent stretch reflex. This interdependence of muscle lengthening and stretch reflexes may be important to consider when administering treatment. However, muscle and tendon lengthening properties alone could not fully explain the variability in stretch reflexes, indicating that other factors should also be investigated.

Muscle spasticity is characterized by exaggerated stretch reflexes and affects about 85% of the children with cerebral palsy. However, the mechanisms underlying spasticity and its influence on gait are not well understood. Here, we first aimed to model the response of spastic hamstrings and gastrocnemii in children with cerebral palsy to fast passive stretches. Then, we evaluated how the model applied to gait. We developed three models based on exaggerated proprioceptive feedback. The first model relied on feedback from muscle fiber length and velocity (velocity-related model), the second model relied on feedback from muscle fiber length, velocity, and acceleration (acceleration-related model), and the third model relied on feedback from muscle force and its first time derivative (force-related model). The force-related model better reproduced measured hamstrings and gastrocnemii activity during fast passive stretches (coefficients of determination (R2): 0.73 ± 0.10 and 0.60 ± 0.13, respectively, and root mean square errors (RMSE): 0.034 ± 0.031 and 0.009 ± 0.007, respectively) than the velocity-related model (R2: 0.46 ± 0.15 and 0.07 ± 0.13, and RMSE: 0.053 ± 0.051 and 0.015 ± 0.009), and the acceleration-related model (R2: 0.47 ± 0.15 and 0.09 ± 0.14, and RMSE: 0.052 ± 0.050 and 0.015 ± 0.008). Additionally, the force-related model predicted hamstrings and gastrocnemii activity that better correlated with measured activity during gait (cross correlations: 0.82 ± 0.09 and 0.85 ± 0.06, respectively) than the activity predicted by the velocity-related model (cross correlations: 0.49 ± 0.17 and 0.71 ± 0.22) and the acceleration-related model (cross correlations: 0.51 ± 0.16 and 0.67 ± 0.20). Our results therefore suggest that force encoding in muscle spindles in combination with altered feedback gains and thresholds underlie activity of spastic muscles during passive stretches and gait. Our model of spasticity opens new perspectives for studying movement impairments due to spasticity through simulation.

The main goal of this validation study was to evaluate whether lower limb muscle weakness and plantar flexor rate of force development (RFD) related to altered gait parameters in children with cerebral palsy (CP), when weakness was assessed with maximal voluntary isometric contractions (MVICs) in a gait related test position. As a subgoal, we analyzed intra- and intertester reliability of this new strength measurement method.Part 1 -Intra- and intertester reliability were determined with the intra-class correlation coefficient (ICC2,1) in 10 typical developing (TD) children (age: 5-15). We collected MVICs in four lower limb muscle groups to define maximum joint torques, as well as plantar flexor RFD. Part 2 -Validity of the strength assessment was explored by analyzing the relations of lower limb joint torques and RFD to a series of kinematic- and kinetic gait features, the GDI (gait deviation index), and the GDI-kinetic in 23 children with CP (GMFCS I-II; age: 5-15) and 23 TD children (age: 5-15) with Spearman's rank correlation coefficients.Part 1 -The best reliability was found for the torque data (Nm), with the highest ICC2,1 (0.951) for knee extension strength (inter) and the lowest (0.693) for dorsiflexion strength (intra). For plantar flexor RFD, the most reliable window size was 300 milliseconds (ICC2,1: 0.828 (inter) and 0.692 (intra)). Part 2 -The children with CP were significantly weaker than the TD children (p

Cerebral palsy (CP) and Duchenne muscular dystrophy (DMD) are neuromuscular disorders characterized by muscle weakness. Weakness in CP has neural and non-neural components, whereas in DMD, weakness can be considered as a predominantly non-neural problem. Despite the different underlying causes, weakness is a constraint for the central nervous system when controlling gait. CP demonstrates decreased complexity of motor control during gait from muscle synergy analysis, which is reflected by a higher total variance accounted for by one synergy (tVAF1). However, it remains unclear if weakness directly contributes to higher tVAF1 in CP, or whether altered tVAF1 reflects mainly neural impairments. If muscle weakness directly contributes to higher tVAF1, then tVAF1 should also be increased in DMD. To examine the etiology of increased tVAF1, muscle activity data of gluteus medius, rectus femoris, medial hamstrings, medial gastrocnemius, and tibialis anterior were measured at self-selected walking speed, and strength data from knee extensors, knee flexors, dorsiflexors and plantar flexors, were analyzed in 15 children with CP [median (IQR) age: 8.9 (2.2)], 15 boys with DMD [8.7 (3.1)], and 15 typical developing (TD) children [8.6 (2.7)]. We computed tVAF1 from 10 concatenated steps with non-negative matrix factorization, and compared tVAF1 between the three groups with a Mann-Whiney U-test. Spearman's rank correlation coefficients were used to determine if weakness in specific muscle groups contributed to altered tVAF1. No significant differences in tVAF1 were found between DMD [tVAF1: 0.60 (0.07)] and TD children [0.65 (0.07)], while tVAF1 was significantly higher in CP [(0.74 (0.09)] than in the other groups (both p < 0.005). In CP, weakness in the plantar flexors was related to higher tVAF1 (r = −0.72). In DMD, knee extensor weakness related to increased tVAF1 (r = −0.50). These results suggest that the non-neural weakness in DMD had limited influence on complexity of motor control during gait and that the higher tVAF1 in children with CP is mainly related to neural impairments caused by the brain lesion.

BACKGROUND AND AIM:Understanding the maturation of upper limb (UL) movement characteristics in typically developing (TD) children is key to explore UL deficits in those with neurodevelopmental disorders. Three-dimensional motion analysis (3DMA) offers a reliable tool to comprehensively evaluate UL motion. However, studies thus far mainly focused on specific pre-defined parameters extracted from kinematic waveforms. Here, we investigated age-related differences in UL movement characteristics over the entire movement cycle in TD children. PARTICIPANTS AND METHODS:We assessed the non-dominant UL of 60 TD children (mean age 10y3m±3y1m) using 3DMA during eight tasks: reaching (forwards (RF), upwards (RU), sideways (RS)), reach-to-grasp (sphere (RGS), vertical cylinder (RGV)) and activities-of-daily-living mimicking tasks (hand-to-head (HTH), hand-to-mouth (HTM), hand-to-shoulder (HTS)). We investigated differences between four age-groups (5-7y, 8-10y, 11-12y, 13-15y) in: (1) spatiotemporal parameters (movement duration, peak velocity, time-to-peak velocity and trajectory straightness), and (2) 12 UL joint angles, using Statistical Parametric Mapping (SPM). RESULTS:We found that the 5-7y children moved with lower peak velocity and less straight trajectories compared to the 11-12y group (peak velocity: RS, HTS, p

Background: In children with unilateral cerebral palsy (uCP) virtually nothing is known on the relation between structural brain damage and upper limb (UL) kinematics quantified with three-dimensional movement analysis (3DMA). This explorative study aimed to (1) investigate differences in UL kinematics between children with different lesion timings, i.e., periventricular white matter (PWM) vs. cortical and deep gray matter (CDGM) lesions and (2) to explore the relation between UL kinematics and lesion location and extent within each lesion timing group.Methods: Forty-eight children (age 10.4 ± 2.7 year; 29 boys; 21 right-sided; 33 PWM; 15 CDGM) underwent an UL 3DMA during a reach-to-grasp task. Spatiotemporal parameters [movement duration, (timing of) maximum velocity, trajectory straightness], the Arm Profile Score (APS) and Arm Variable Scores (AVS) were extracted. The APS and AVS refer to the total amount of movement pathology and movement deviations of the wrist, elbow, shoulder, scapula and trunk respectively. Brain lesion location and extent were scored based on FLAIR-images using a semi-quantitative MRI-scale.Results: Children with CDGM lesions showed more aberrant spatiotemporal parameters (p < 0.03) and more movement pathology (APS, p = 0.003) compared to the PWM group, mostly characterized by increased wrist flexion (p = 0.01). In the CDGM group, moderate to high correlations were found between lesion location and extent and duration, timing of maximum velocity and trajectory straightness (r = 0.53–0.90). Lesion location and extent were further moderately correlated with distal UL movement pathology (wrist flexion/extension, elbow pronation/supination, elbow flexion/extension; r = 0.50–0.65) and with the APS (r = 0.51–0.63). In the PWM group, only a few and low correlations were observed, mostly between damage to the PLIC and higher AVS of elbow flexion/extension, shoulder elevation and trunk rotation (r = 0.35–0.42). Regression analysis revealed damage to the temporal lobe with lesion timing as interactor (27%, p = 0.002) and the posterior limb of the internal capsule (PLIC) (7%, p = 0.04) as the strongest predictors, explaining 34% of the variance in APS.Conclusion: UL kinematic deviations are more influenced by lesion location and extent in children with later (CDGM) versus earlier lesions (PWM), except for proximal movement pathology. Damage to the PLIC is a significant predictor for UL movement pathology irrespective of lesion timing.

Upper limb three-dimensional movement analysis (UL-3DMA) offers a reliable and valid tool to evaluate movement patterns in children with unilateral cerebral palsy (uCP). However, it remains unknown to what extent the underlying motor impairments explain deviant movement patterns. Such understanding is key to develop efficient rehabilitation programs. Although UL-3DMA has been shown to be a useful tool to assess movement patterns, it results in a multitude of data, challenging the clinical interpretation and consequently its implementation. UL-3DMA reports are often reduced to summary metrics, such as average or peak values per joint. However, these metrics do not take into account the continuous nature of the data or the interdependency between UL joints, and do not provide phase-specific information of the movement pattern. Moreover, summary metrics may not be sensitive enough to estimate the impact of motor impairments. Recently, Statistical Parametric Mapping (SPM) was proposed to overcome these problems. We collected UL-3DMA of 60 children with uCP and 60 typically developing children during eight functional tasks and evaluated the impact of spasticity and muscle weakness on UL movement patterns. SPM vector field analysis was used to analyze movement patterns at the level of five joints (wrist, elbow, shoulder, scapula, and trunk). Children with uCP showed deviant movement patterns in all joints during a large percentage of the movement cycle. Spasticity and muscle weakness negatively impacted on UL movement patterns during all tasks, which resulted in increased wrist flexion, elbow pronation and flexion, increased shoulder external rotation, decreased shoulder elevation with a preference for movement in the frontal plane and increased trunk internal rotation. Scapular position was altered during movement initiation, although scapular movements were not affected by muscle weakness or spasticity. In conclusion, we identified pathological movement patterns in children with uCP and additionally mapped the negative impact of spasticity and muscle weakness on these movement patterns, providing useful insights that will contribute to treatment planning. Last, we also identified a subset of the most relevant tasks for studying UL movements in children with uCP, which will facilitate the interpretation of UL-3DMA data and undoubtedly contribute to its clinical implementation.

Elastic tape has been widely used in clinical practice in order to improve upper limb (UL) sensibility. However, there is little evidence that supports this type of intervention in stroke patients.To verify the effect of elastic tape, applied to the paretic shoulder, on joint position sense (JPS) during abduction and flexion in subjects with chronic hemiparesis compared to sham tape (non-elastic tape). Furthermore, to verify if this potential effect is correlated to shoulder subluxation measurements and sensorimotor impairment.A crossover and sham-controlled study was conducted with post-stroke patients who were randomly allocated into two groups: 1) those who received Sham Tape (ST) first and after one month they received Elastic Tape (ET); 2) those who received Elastic Tape (ET) first and after one month they received Sham Tape (ST). The JPS was evaluated using a dynamometer. The absolute error for shoulder abduction and flexion at 30° and 60° was calculated. Sensorimotor impairment was determined by Fugl-Meyer, and shoulder subluxation was measured using a caliper.Thirteen hemiparetic subjects (average time since stroke 75.23 months) participated in the study. At baseline (before interventions), the groups were not different for abduction at 30° (p = 0.805; p = 0.951), and 60° (p = 0.509; p = 0.799), or flexion at 30° (p = 0.872; p = 0.897) and 60° (p = 0.853; p = 0.970). For the ET group, differences between pre and post-elastic tape for abduction at 30° (p