PURPOSE:To analyze the effect of home-based therapy for upper limb activity in children and adolescents with unilateral cerebral palsy. METHODS:PubMed, Cochrane Central Register of Controlled Trials, CINAHL, PsycINFO, and PEDro were systematically searched from inception to March 2026 for experimental studies addressing the effect of home-based therapies in participants aged from 2 to 18 years old. The DerSimonian and Laird method was used to compute pooled estimates of the standardized mean differences (SMD) and their respective 95% confidence intervals (CI). Subgroup analyses were performed based on the dosage of interventions. RESULTS:Seventeen studies were included in this systematic review. Home-based therapies showed improvement in upper limb activity (SMD = 0.45; 95% CI: 0.30-0.60). No statistically significant differences were observed in relation to the intensity of intervention (hours per week). Meta-regression analysis revealed that none of the implementation-related moderators-including parental involvement, delivery format, dose profile, task relevance and contextual support-had a statistically significant effect on intervention outcomes (p > 0.05). CONCLUSIONS:High-intensity home-based therapies have demonstrated promising findings in improving upper limb activity in children and adolescents with unilateral cerebral palsy. Further high-quality studies may assess the influence of intervention characteristics.

AIM Executive functions (EF) are advanced cognitive processes that play an essential role in daily functioning and may be of increased importance in cerebral palsy (CP), given the complexity of primary and associated impairments. This study aims to synthesize existing evidence on the relation between EF and domains of the International Classification of Functioning, Disability and Health (ICF) in individuals with CP, and to quantify the magnitude of these associations through meta-analysis. METHOD A systematic literature search was conducted in eight electronic databases up to 14 July 2025, examining associations between EF and ICF domains in CP. EF outcomes were classified into inhibitory control, working memory, cognitive flexibility, higher-order EF, and EF composite scores. Outcome measures were mapped onto ICF domains: Body Functions and Structures, Activity, Participation, and Contextual factors, using the CP Core Sets. Correlation coefficients were transformed to Fisher's z and entered into three-level meta-analyses to estimate pooled effect sizes. Single moderator analyses examined CP subtype, EF domain, EF assessment type, and mean age. Risk of bias was assessed using the Quality in Prognosis Studies (QUIPS) tool. RESULTS From 4637 identified records, 38 studies were included, comprising a total sample of 1633 participants with CP. There was substantial heterogeneity in CP subtype, participant age, and EF conceptualization, while the ICF Contextual factors domain was underrepresented. A medium-to-large association was found between EF and functioning across all ICF domains combined (r=0.26, p<0.001). Domain-specific analyses showed a medium association of EF with Body Functions and Structures (r=0.21, p<0.01), a medium-to-large association with Activity (r=0.38, p<0.001) and Participation (r=0.26, p<0.01). CP subtype and mean age significantly moderated the overall EF-functioning association, with mixed CP and younger age associated with stronger effects. INTERPRETATION EF are meaningfully associated with multiple domains of functioning in individuals with CP. These findings support the relevance of routine EF assessment and suggest that EF are an important cognitive correlate to consider when addressing broader aspects of daily functioning. ### Competing Interest Statement The authors have declared no competing interest. ### Clinical Protocols ### Funding Statement Alexandra Kalkantzi as first author was supported by funding from the European Commission, Horizon Europe Research and Innovation Action under Grant Agreement No 101057309, in the context of the AINCP project. Saranda Bekteshi as last author was supported by FWO junior postdoctoral fellowship, project nr 1264123N. The work for Giuseppina Sgandurra was also partially supported by the Italian Ministry of Health Grant RC2025 (and the 5x1000 voluntary contributions). The funders had no role in the design of the systematic review or in the drafting of the review protocol or manuscript. ### Author Declarations I confirm all relevant ethical guidelines have been followed, and any necessary IRB and/or ethics committee approvals have been obtained. Yes The details of the IRB/oversight body that provided approval or exemption for the research described are given below: This study comprises a systematic review and meta-analysis, therefore, all included data derive from already published clinical studies. The included studies are clearly stated inside the manuscript. I confirm that all necessary patient/participant consent has been obtained and the appropriate institutional forms have been archived, and that any patient/participant/sample identifiers included were not known to anyone (e.g., hospital staff, patients or participants themselves) outside the research group so cannot be used to identify individuals. Yes I understand that all clinical trials and any other prospective interventional studies must be registered with an ICMJE-approved registry, such as ClinicalTrials.gov. I confirm that any such study reported in the manuscript has been registered and the trial registration ID is provided (note: if posting a prospective study registered retrospectively, please provide a statement in the trial ID field explaining why the study was not registered in advance). Yes I have followed all appropriate research reporting guidelines, such as any relevant EQUATOR Network research reporting checklist(s) and other pertinent material, if applicable. Yes All data produced in the present study are available upon reasonable request to the authors

BACKGROUND:In children with unilateral cerebral palsy (uCP), the relation between different factors (brain damage, prematurity, and cerebral palsy side) and visual impairments, which are common but often 'unseen,' is not fully understood. METHODS:Visual functions and functional vision were assessed in 41 children with uCP (7-15 years, 21 left-sided, 19 preterm). Brain damage was scored on structural magnetic resonance imaging regarding lesion timing, location, and severity, and corpus callosum (CC) length and splenium thickness. With nonparametric statistics, we investigated the relation between visual outcomes and brain damage (rs) and differences in visual outcomes and brain damage based on prematurity and uCP side (r). With elastic-net regularized regression models (area under the receiver operating characteristic curve [AUC]; d), we explored if gestational age and brain damage could predict impairments in visual functions. RESULTS:Damage to the lobes and CC was associated with reduced visual functions and functional vision (rs = -0.402 to -0.611). Compared to children born at term, preterm children mainly showed reduced geniculostriate functions (r = 0.343-0.443) and damage to the parietal lobe (r = 0.353). No differences in brain damage were found between children with left- and right-sided uCP. In regression models, shorter CC length and parietal lobe lesion (d = -0.526 to 0.564) were the main predictors for impaired stereoacuity (AUC = 0.77), and occipital lobe lesion (d = 0.349) for impaired visuomotor integration (AUC = 0.81). Prediction models of visual perception showed poor predictive performance (AUC<0.70). CONCLUSIONS:Specific brain damage and prematurity are related to different visual impairments in children with uCP. Our results could guide clinicians in directing their attention to specific visual assessments and subsequent intervention in children with uCP.

Children with unilateral cerebral palsy (uCP) frequently experience bimanual coordination difficulties. Executive functions (EFs), which support goal-directed behavior, may be associated with these difficulties. This study investigated the relation between daily-life EFs and goal-directed bimanual coordination in children with uCP by examining the effects of EFs and age on coordination, and associations between EF domains and bimanual coordination. Bimanual coordination was assessed in 46 children with uCP (mean age 11.1 ± 2.1 years) and 56 typically developing children (mean age 11.7 ± 2.8 years), through three tasks of the Kinarm exoskeleton involving target reaching, ball interception, circuit navigation. Data from typically developing children were used to establish age-predictive norms for bimanual coordination. Daily-life EFs in children with uCP were assessed using the Behavior Rating Inventory of Executive Function, classifying them as typical EFs (n = 21) and below-average EFs (n = 25). Group differences in impairments were examined using Fisher’s exact tests, while multiple linear regressions estimated the effects of EF group and age on bimanual coordination; sensitivity analyses excluding children with co-occurring conditions were additionally performed for these models. Spearman correlations (rₛ) explored associations between daily-life EFs and bimanual coordination. Children with below-average EFs showed more bimanual coordination impairments in seven parameters (p ≤ 0.045). Sensitivity analyses showed similar patterns. EF group effects were found for five bimanual parameters (R² up to 0.24), favoring the typical EF group; two parameters improved with increasing age (R² up to 0.30). Low-to-moderate correlations were found between daily-life EFs and bimanual coordination (rₛ up to 0.56). These findings suggest that daily-life EFs are associated with bimanual coordination variability in uCP. Clinically, they highlight the importance of considering cognitive abilities during assessment and incorporating cognitive demands within bimanual rehabilitation approaches in uCP.

PURPOSE:To map somatosensory impairments in the more-impaired and less-impaired upper limb of children with unilateral cerebral palsy (uCP) compared to typically developing children (TDC), using clinical and robotic assessments. Additionally, we explored which subgroups of children with uCP are more likely to experience somatosensory impairments. MATERIALS AND METHODS:This cross-sectional study included 49 children with uCP (11y11m ± 2y10m, 26 males) and 49 age- and sex-matched TDC (11y10m ± 2y10m). Tactile registration (Semmes-Weinstein monofilaments), tactile perception (stereognosis, two-point discrimination), and proprioception (clinical movement sense, ETH MIKE robot, and Kinarm exoskeleton) were assessed. Differences between groups and across manual ability levels and brain lesion types were analyzed using generalized estimating equation models. RESULTS:Children with uCP exhibited tactile impairments in both hands (more-impaired: 27-43%; less-impaired: 12-16%) and worse proprioception bilaterally compared to TDC (p ≤ 0.003). Lower manual ability was related to worse tactile perception (p ≤ 0.001) and proximal proprioception (p ≤ 0.045). Predominant grey matter lesions were correlated with worse tactile registration (p = 0.016), perception (p ≤ 0.009), and distal proprioception (p = 0.009). CONCLUSIONS:Our results underline the importance of including clinical and robotic assessments in the follow-up of children with uCP to identify bilateral somatosensory impairments. An international consensus on clinically relevant and well-defined assessment protocols is needed to enhance clinical translation.

Optimal upper limb (UpL) function is essential for performing daily activities; however, children with unilateral spastic cerebral palsy (USCP) often experience impairments in UpL function, which can impact their quality of life or independence. While UpL motor impairments are a primary concern, non-motor functions, such as cognition, attention, and visual function, commonly impaired in USCP, may also play a role in UpL performance. Nevertheless, these non-motor functions are often not considered in evaluation protocols that focus on the UpL. Moreover, clinical evaluation is typically conducted in structured and controlled settings and may not accurately reflect the child’s abilities in daily life. Non-invasive, novel technologies are a promising solution for filling this gap, by providing additional quantitative and ecologically valid information to clinicians. In this context, this overview aims (i) to present the most frequently used tools for a holistic evaluation in children with USCP, ensuring a thorough understanding of the UpL function, and (ii) to report the evidence of how novel, non-invasive technologies can enhance clinical evaluation in daily life, enabling a more comprehensive evaluation. This work could set a basis for multidimensional evaluation protocols for UpL function in USCP, providing a different approach to the current standards.

Children with unilateral cerebral palsy (uCP) present with brain damage, predominantly lateralized to one hemisphere, and white matter (WM) lesions, which are known to affect visual functions. However, the relation between WM tract damage and visual outcomes remains unclear. Additionally, no prior study comprehensively investigated hemispheric-specific differences in WM visual pathways between children with left- and right-sided uCP. Therefore, this exploratory study aims to investigate differences in micro- and macrostructural properties of the visual pathways between children with left- and right-sided uCP and their relation to visual outcomes, using fixel-based analysis of diffusion MRI (dMRI). dMRI data and visual assessments, including visual acuity and stereoacuity (i.e., geniculostriate functions), motor-free visual perception, visuomotor integration, and functional vision, were analysed in 36 children with uCP (aged 7-15, 9 males, 17 left-sided, 15 preterm). Apparent fiber density (AFD), fiber-bundle cross-section (FC), and combined fiber density and cross-section (FDC) were calculated for 17 WM tracts related to visual functions. Differences between children with left- and right-sided uCP were investigated using the Mann-Whitney U-test (r) on the AFD and one-way analysis of covariance (ANCOVA) (ηp 2 ) on the FC and FDC, with age and intracranial volume as covariates. Correlations between visual outcomes and WM properties of the visual tracts were studied using (semi-partial) Spearman Rank correlations (rs ). Children with left-sided uCP showed significantly lower fixel metrics in the right superior longitudinal fasciculus, inferior fronto-occipital fasciculus, and optic radiation. Children with right-sided uCP had lower AFD, FC, and FDC in the left superior longitudinal fasciculus only. Reduced geniculostriate visual functions and more impairments in functional vision were associated with lower fiber density (AFD), reduction in bundle size (FC), and their combination (FDC) of several WM tracts. Lower performance on motor-free visual perception and visuomotor integration showed more associations with lower fiber density (AFD). While the primary analyses were exploratory and uncorrected for multiple comparison, false discovery rate (FDR) correction was additionally performed for transparency: several differences in FC and FDC between children with left- and right-sided uCP, and correlations between AFD and visual function, remained significant and are reported in the Supplementary Materials. In conclusion, our exploratory study highlights that fixel-based analysis can provide further insights into hemispheric differences in the visual system and the complex relations between visual functions and brain damage in children with uCP. Based on our results, future studies could refine regression models to target key WM tracts linked to visual outcomes, identifying potential biomarkers to predict visual impairments and enable early tailored support in children with uCP.

AIM:To explore daily-life reported executive functions and their relation with bimanual performance in children with unilateral cerebral palsy (CP). METHOD:In this cross-sectional study of 46 children with unilateral CP (mean age 11 years 10 months, standard deviation 2 years 10 months), executive functions were evaluated using the Behavior Rating Inventory of Executive Function (BRIEF) and bimanual performance with the Assisting Hand Assessment (AHA) and Children's Hand-use Experience Questionnaire (CHEQ). One-sample z-tests were used to compare participants' executive functions with population norms, while taking autism spectrum disorder (ASD, n = 16) as a comorbidity into account. Moreover, we used regression analysis to estimate the effect of manual ability (Manual Ability Classification System levels: I = 25, II = 15, III = 6) and having a comorbid diagnosis of ASD on executive functions (p < 0.05, R2). Lastly, non-parametric correlations (rs, p < 0.05) were calculated between the BRIEF, CHEQ, and AHA. RESULTS:In general, executive functions in children with unilateral CP were poorer compared with the normative mean (p ≤ 0.024). However, when excluding participants with ASD, no difference compared with the normative mean was found. A significant effect of manual ability was found for Inhibition (p = 0.042), while ASD effects were found for most of the BRIEF subscales (p ≤ 0.001). Multiple significant correlations were found between the BRIEF and CHEQ (rs = -0.50 to -0.29), while only the BRIEF subscale Inhibition was significantly correlated with the AHA (rs = -0.35). INTERPRETATION:A higher number of children with unilateral CP exhibit difficulties in daily-life executive functions, which appear to be mainly co-occurring with ASD. Manual ability was a significant factor of inhibition-related behavioural challenges. Furthermore, there seems to be a relation between impaired executive functions and decreased bimanual performance. The findings emphasize the importance of further research, including performance-based assessments of executive functions in children with unilateral CP.

Poor manual skills in children with Developmental Coordination Disorder (DCD) may be dependent on task complexity and due to difficulties in the automatization phase of the motor learning process. Increased task demands and the dual-task paradigm can be used to test these hypotheses. OBJECTIVES:We aim to investigate (1) manual dexterity abilities using increased levels of difficulty; (2) dual tasking using an experimental protocol of the Tyneside Pegboard Test (TPT). METHOD:Sixteen children with DCD and 16 age-matched typically developing (TD) children were included. Various experimental conditions of the TPT (unimanual, bimanual and dual task) were administered. The dual-task paradigm comprised a primary unimanual task and a cognitive task (auditory non-verbal task). Parents were asked to fill out the eConners questionnaire to report attentional difficulties. Repeated measures ANOVAs were used to compare possible differences in effects on the performance of the groups. Pearson correlation coefficients were calculated between dual-task performance and ADHD index of the eConners questionnaire. RESULTS:Children with DCD performed significantly worse in all task conditions (unimanual, bimanual, dual task) compared to TD children. In unimanual and bimanual conditions, they did no not present a higher impact of task constraints. Dual-task performances did not have a differential effect on groups and were not interfered by attentional difficulties. CONCLUSIONS:Children with DCD exhibit a general slowness in all TPT tasks. Our findings do not support the automatization deficit hypothesis.

Background: In children with unilateral cerebral palsy (uCP), bimanual assessments mostly focus on qualitative assessments of the impaired upper limb during bimanual tasks, which do not capture the spatiotemporal coordination between both hands. Hence, we aimed to advance our understandings in spatiotemporal coordination in children with uCP compared to typically developing children (TDC) using a bimanual, asymmetrical, goal-directed task. Participants and methodology: In this observational study, thirty-seven children with uCP (11y8m +/- 2y10m, 20 males, 16 right-sided uCP, Manual Ability Classification System level I = 23, II = 11, III = 3) and 37 age and sex-matched TDC opened a box with one hand and pressed a button inside using the opposite hand. Spatiotemporal bimanual (movement time, temporal coupling, movement overlap, goal synchronisation) and unimanual (movement time, path length and smoothness) parameters were extracted. Between groups comparisons were investigated using a two-way mixed ANCOVA with age as covariate (alpha < 0.05). Additionally, correlation coefficients between unimanual and bimanual parameters were calculated. Results: Compared to TDC, children with uCP were slower (p = 0.01, eta(2)(p) = 0.13) and presented unimanual spatiotemporal deficits in both upper limbs (p < 0.03, eta(2)(p)>0.10), which worsened in children with lower manual abilities (p < 0.04, eta(2)(p)>0.19). However, they did not differ in bimanual coupling (p > 0.31, eta(2)(p)<0.03). Furthermore, slower movement time was related with increased unimanual spatiotemporal deficits bilaterally (r = 0.34-0.80, p = 0.001-0.04), suggesting that reduced performance at both upper limbs contributes to bimanual difficulties in children with uCP. Conclusions: The bilateral reduced spatiotemporal performance, related to longer bimanual movement time, stresses the importance to assess and treat both upper limbs in children with uCP.

ABSTRACT Transcranial direct current stimulation (tDCS) may facilitate neuroplasticity but with a limited effect when administered while stroke patients are at rest. Muscle-computer interface (MCI) training is a promising approach for training stroke patients even if they cannot produce overt movements. However, using tDCS to enhance MCI training has not been investigated. We combined bihemispheric tDCS with MCI training of the paretic wrist and examined the effectiveness of this intervention in chronic stroke patients. A crossover, double-blind, randomized trial was conducted. Twenty-six chronic stroke patients performed MCI wrist training for three consecutive days at home while receiving either real tDCS or sham tDCS in counterbalanced order and separated by at least 8 months. The primary outcome measure was the Fugl-Meyer Assessment Upper Extremity Scale (FMA-UE) which was measured one week before training, on the first training day, on the last training day, and one week after training. There was no significant difference in the baseline FMA-UE score between groups nor between intervention periods. Patients improved 3.850 ± 0.582 points in FMA-UE score when receiving real tDCS, and 0.963 ± 0.725 points when receiving sham tDCS (p=0.003). Additionally, patients also showed continuous improvement of their motor control of the MCI tasks over the training days. Our study showed that the training paradigm could lead to functional improvement in chronic stroke patients. We argue that an appropriate MCI training in combination with bihemispheric tDCS could be a useful adjuvant for neurorehabilitation in stroke patients. NEW & NOTEWORTHY Bihemispheric tDCS combined with a novel MCI training for motor control of wrist extensor can improve both proximal and distal arm function in chronic stroke patients. The training regimen can be personalized with adjustments made daily to accommodate the functional change throughout the intervention. This demonstrates that bihemispheric tDCS with MCI training could complement conventional post-stroke neurorehabilitation.

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

BACKGROUND:Boys with Duchenne Muscular Dystrophy (DMD) experience both fine and gross motor problems. Nowadays, early intervention focuses almost exclusively on gross motor skills. AIMS:We aimed to explore early motor development in preschool boys with DMD and investigate the influence of cognition. METHODS AND PROCEDURES:Seventeen boys with DMD (11 months- 6 years) were compared to typically developing (TD) peers and followed-up with the Bayley Scales of Infant and Toddler Development (Bayley-III); Peabody developmental motor scales (PDMS-II) and Motor Function Measure (MFM-20). The longitudinal evolution of fine and gross motor skills was investigated using linear mixed effect models (LMM). Cognition was added to the LMM as a covariate. OUTCOMES AND RESULTS:Preschool boys with DMD scored lower compared to TD peers on both fine and gross motor skills (p<0.001). The evolution of motor development was subscale-dependent. A significant influence of cognition was found on different subscales (p= 0.002-0.04). CONCLUSIONS AND IMPLICATIONS:Preschool boys with DMD do not achieve the same functioning level as TD boys. Cognition plays a crucial role in the evolution of motor skills. Our results suggest a shift to a broader psychomotor approach including both fine and gross motor skills, also considering the impact of cognition. WHAT THIS PAPER ADDS?: Our study provides a detailed mapping of early fine and gross motor development in preschool boys with Duchenne Muscular Dystrophy (DMD) and describes the influence of cognition on both fine and gross motor skills. Preschool boys with DMD do not achieve the same functioning level compared to typically developing boys. They score significantly lower on both fine and gross motor skills. The evolution of fine and gross motor development was subscale-dependent e.g. a negative-positive evolution was seen for grasping skills, with a tipping point around the age of four; stationary scaled scores decreased followed by a stabilization around the age four to five and locomotion scaled scores remained stable over time. Finally, we also found that cognition plays a crucial role in the evolution of both fine and gross motor skills. These new insights in the evolution of early motor development could be of added value for future clinical trials in young boys with DMD. Subsequently, increased alertness to early symptoms, e.g. developmental delay, may advance the age of diagnosis, as well as associated early intervention.

Background: Visual information is crucial for performing goal-directed movements in daily life. Aims: To investigate the relation between visual functions, functional vision, and bimanual function in children with unilateral cerebral palsy (uCP). Methods and procedures: In 49 children with uCP (7-15y), we investigated the relation between stereoacuity (Titmus Stereo Fly test), visual perception (Test of Visual Perceptual Skills), visuomotor integration (Beery Buktenica Test of Visual-Motor Integration) and functional vision (Flemish cerebral visual impairment questionnaire) with bimanual dexterity (Tyneside Pegboard Test), bimanual coordination (Kinarm exoskeleton robot, Box opening task), and functional hand use (Children’s Hand-use Experience Questionnaire; Assisting Hand Assessment) using correlations ( r s ) and elastic-net regularized regressions ( d ). Outcomes and results: Visual perception correlated with bimanual coordination ( r s =0.407-0.436) and functional hand use ( r s =0.380-0.533). Stereoacuity ( r s =-0.404), visual perception ( r s =-0.391-(-0.620)), and visuomotor integration ( r s =-0.377) correlated with bimanual dexterity. Functional vision correlated with functional hand use ( r s =-0.441-(-0.458)). Visual perception predicted bimanual dexterity ( d =0.001-0.315), bimanual coordination ( d =0.004-0.176), and functional hand use ( d =0.001-0.345), whereas functional vision mainly predicted functional hand use ( d =0.001-0.201). Conclusions and implications: Visual functions and functional vision are related to bimanual function in children with uCP highlighting the importance of performing extensive visual assessment to better understand children's difficulties in performing bimanual tasks.

Background Besides motor impairments, up to 90% of the children and adolescents with unilateral cerebral palsy (uCP) present with somatosensory impairments in the upper limb. As somatosensory information is of utmost importance for coordinated movements and motor learning, somatosensory impairments can further compromise the effective use of the impaired upper limb in daily life activities. Yet, intervention approaches specifically designated to target these somatosensory impairments are insufficiently investigated in children and adolescents with uCP. Therefore, the aim of this randomized controlled trial (RCT) is to compare the effectiveness of somatosensory discrimination therapy and dose-matched motor therapy to improve sensorimotor upper limb function in children and adolescents with uCP, who experience somatosensory impairments in the upper limb. We will further explore potential behavioral and neurological predictors of therapy response. Methods A parallel group, evaluator-blinded, phase-II, single-center RCT will be conducted for which 50 children and adolescents with uCP, aged 7 to 15 years, will be recruited. Participants will be randomized to receive 3 weekly sessions of 45 minutes of either somatosensory discrimination therapy or upper limb motor therapy for a period of 8 weeks. Stratification will be performed based on age, manual ability, and severity of tactile impairment at baseline. Sensorimotor upper limb function will be evaluated at baseline, immediately after the intervention and after 6 months follow-up. The primary outcome measure will be bimanual performance as measured with the Assisting Hand Assessment. Secondary outcomes include a comprehensive test battery to objectify somatosensory function and measures of bimanual coordination, unimanual motor function, and goal attainment. Brain imaging will be performed at baseline to investigate structural brain lesion characteristics and structural connectivity of the white matter tracts. Discussion This protocol describes the design of an RCT comparing the effectiveness of somatosensory discrimination therapy and dose-matched motor therapy to improve sensorimotor upper limb function in children and adolescents with uCP. The results of this study may aid in the selection of the most effective upper limb therapy, specifically for children and adolescents with tactile impairments. Trial registration ClinicalTrials.gov (NCT06006065). Registered on August 8, 2023.

Duchenne Muscular Dystrophy (DMD) is an X-linked recessive disorder caused by mutations in the dystrophin gene. Deficiency of the dystrophin protein causes not only motor, but also cognitive, language, behavioural and social emotional problems. This is the first systematic review investigating five early developmental domains in boys with DMD between 0 and 6 years old. Interactions between different domains and links with mutation types and sites were explored.A systematic search was performed in PubMed, Web of Science and Scopus. An adapted version of the Scottish Intercollegiate Guidelines Network (SIGN) Checklists for case-control and cohort studies was used to evaluate quality.Fifty-five studies of high or acceptable quality were included. One was an RCT of level 1b; 50 were cohort studies of level 2b; and four were an aggregation of case-control and cohort studies receiving levels 2b and 3b. We found that young boys with DMD experienced problems in all five developmental domains, with significant interactions between these. Several studies also showed relationships between mutation sites and outcomes.We conclude that DMD is not only characterised by motor problems but by a more global developmental delay with a large variability between boys. Our results emphasise the need for harmonisation in evaluation and follow-up of young boys with DMD. More high-quality research is needed on the different early developmental domains in young DMD to facilitate early detection of difficulties and identification of associated early intervention strategies.

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

This commentary is on the original article by Hoskens et al. on pages 644–653 of this issue.