Multisensory Stimulation and Priming (MuSSAP) in 4-10 Months Old Infants with a Unilateral Brain Lesion: A Pilot Randomised Clinical Trial.

Aim: To explore the effect of an Early Intensive-Upper Limb intervention (EI-UL) compared to EI-UL with integrated Multisensory Stimulation And Priming (MuSSAP) training on improving manual ability in infants with a unilateral brain lesion. Method: A pilot randomised clinical trial with pre- and postintervention and follow-up measurements (T0, T1, and T2) was conducted. Sixteen infants with a unilateral brain lesion (corrected age is 4-10 months) received home-based intervention with video coaching. Eight infants received EI-UL and eight infants received EI-UL with integrated MuSSAP training. Primary outcome was the Hand Assessment for Infants (HAI) score. Additionally, effects were explored on initiation of goal-directed movements in both groups and on attention in the EI-UL with integrated MuSSAP training group. Results: No significant group differences in HAI scores were found. Overall, HAI 'Affected hand score' increased between T0 and T1 (p = 0.001, Cohen's d = 1.04) and between T0 and T2 (p < 0.001, Cohen's d = 1.28); and the HAI 'Both Hands Measure' increased between T0 and T1 (p < 0.001, Cohen's d = 1.72) and between T0 and T2 (p < 0.001, Cohen's d = 1.81). At the start of the intervention, six infants (three in both groups) did not demonstrate initiation of goal-directed contralesional upper limb movements. During the intervention one infant receiving EI-UL and all three infants receiving EI-UL with integrated MuSSAP training started to initiate goal-directed movements. Conclusion: The results suggest manual ability of infants with unilateral brain lesion improved with both interventions. We hypothesize that the integrated MuSSAP training may facilitate attention and initiation of contralesional upper limb goal-directed movements. This trial is registered with NCT05533476).

Parents' experiences with a home-based upper limb training program using a video coaching approach for infants and toddlers with unilateral cerebral palsy: a qualitative interview study.

BACKGROUND: Although early home-based upper limb training programs are promising, in-depth understanding of parents' experiences with these programs is still limited. We developed an early home-based upper limb training program for infants and toddlers (8-36 months) with or at risk of unilateral cerebral palsy using video coaching for parents. The aim of this qualitative study was to evaluate parents' experiences with the home-based training program using a video coaching approach in order to optimize implementation strategies. METHODS: We held semi-structured interviews with parents of 13 children with unilateral cerebral palsy, who participated in our program in the period from 2014 - 2017. On average, parents had delivered two training periods of the program at the time of the interviews. Interviews were analyzed using inductive thematic content analysis. RESULTS: We identified three overarching interacting themes that shaped the experiences of parents with the program: 1) Parental learning comprising the subthemes parents' training competencies and the facilitative and reinforcing role of video coaching, 2) Parental load comprising the subthemes flexibility of the program, supportive network, competing demands, and child's mood and functional capacities, and 3) Parental perseverance comprising the subthemes beliefs and expectancies and seeing child's functional improvements. CONCLUSIONS: For successful implementation of an early home-based upper limb training program using video coaching, support in delivering home-training from a therapist or from others within parents' social network, is needed to relieve parental load. Seeing functional improvements of their child on the videos increased parents' motivation to continue with the training. Positively phrased feedback from an occupational therapist stimulated parents' perseverance and training competency.

Benefits of an Intensive Individual CO-OP Intervention in a Group Setting for Children with DCD.

Purpose: The present study focused on the impact of an adapted Cognitive Orientation to daily Occupational Performance (CO-OP) five-day intervention program for children with developmental coordination disorder (DCD). Important adaptations were the new combination of individual CO-OP sessions and group activities, the short and intensive program that was followed by a training and coaching trajectory, and the use of video logs. Materials and Methods: Eighteen children with DCD (aged 8-16 years) participated in the five-day intervention during which they worked on three intervention goals. After the intervention, during an eight-week training and coaching trajectory for parents and children, children worked on a transfer goal. Assessment took place at four moments in time: two pretest measures, a posttest measure, and a 3-month follow-up measure. Primary outcome measures focused on changes in performance and satisfaction of self-chosen intervention and transfer goals. The secondary outcome measure explored changes in children's attitude, motivation, and confidence in relation to motor skill activities, social skills, and level of participation. Results: Significant improvements were found with regard to the performance and satisfaction of intervention goals. For the transfer goal, only parents reported significant improvements. Finally, parents indicated potential improvements with regard to the attitude, motivation, and confidence of their children, but not for their social skills or level of participation. Conclusion: The findings are promising with regard to the efficacy of this adapted CO-OP intervention for improving intervention goals, but less effective for transfer of learned skills to other goals after the intervention. Future research should focus on how postintervention parental coaching can be improved in order to increase generalization and transfer.

A First Step Toward the Operationalization of the Learned Non-Use Phenomenon: A Delphi Study.

BACKGROUND: The negative discrepancy between residual functional capacity and reduced use of the contralesional hand, frequently observed after a brain lesion, has been termed Learned Non-Use (LNU) and is thought to depend on the interaction of neuronal mechanisms during recovery and learning-dependent mechanisms. OBJECTIVE: Albeit the LNU phenomenon is generally accepted to exist, currently, no transdisciplinary definition exists. Furthermore, although therapeutic approaches are implemented in clinical practice targeting LNU, no standardized diagnostic routine is described in the available literature. Our objective was to reach consensus regarding a definition as well as synthesize knowledge about the current diagnostic procedures. METHODS: We used a structured group communication following the Delphi method among clinical and scientific experts in the field, knowledge from both, the work with patient populations and with animal models. RESULTS: Consensus was reached regarding a transdisciplinary definition of the LNU phenomenon. Furthermore, the mode and strategy of the diagnostic process, as well as the sources of information and outcome parameters relevant for the clinical decision making, were described with a wide range showing the current lack of a consistent universal diagnostic approach. CONCLUSIONS: The need for the development of a structured diagnostic procedure and its implementation into clinical practice is emphasized. Moreover, it exists a striking gap between the prevailing hypotheses regarding the mechanisms underlying the LNU phenomenon and the actual evidence. Therefore, basic research is needed to bridge between bedside and bench and eventually improve clinical decision making and further development of interventional strategies beyond the field of stroke rehabilitation.

The effects of modified constraint-induced movement therapy combined with intensive bimanual training in children with brachial plexus birth injury: a retrospective data base study.

BACKGROUND: In children with Brachial Plexus Birth Injury, a non-use of the affected upper limb despite sufficient capacity, is sometimes observed, called developmental disregard. The combination of modified constraint-induced-movement-therapy with bimanual training is frequently applied to overcome developmental disregard in unilateral Cerebral Palsy. In the current study the effects of the combination of modified constraint-induced-movement-therapy with bimanual training are investigated in children with Brachial Plexus Birth Injury in comparison to children with unilateral Cerebral Palsy. We hypothesize that the combination of modified constraint-induced-movement-therapy with bimanual training is effective in Brachial Plexus Birth Injury. METHODS: Data of 19 children with Brachial Plexus Birth Injury (Mage: 4.1 years) and 18 with unilateral Cerebral Palsy (Mage: 4.5 years) were compared. The effects of modified constraint-induced-movement-therapy with bimanual training (54 h modified constraint-induced-movement-therapy, 18 h bimanual training, 8-10 weeks) was investigated by assessing spontaneous affected-upper-limb-use ("Assisting Hand Assessment"), manual abilities ("ABILHAND-kids") and subjective performance and satisfaction of problematic bimanual activities ("Canadian Occupational Performance Measure") at three time points (pre-treatment, post-treatment, follow-up). This data was analyzed using repeated-measures analysis. RESULTS: Children with Brachial Plexus Birth Injury showed significant improvements on all outcome measures following modified constraint-induced-movement-therapy with bimanual training. These results were comparable to those observed in the group of children with unilateral Cerebral Palsy. DISCUSSION: These results suggest that modified constraint-induced-movement-therapy with bimanual training is effective in Brachial Plexus Birth Injury. They indicate a comparable improved bimanual performance in children with Brachial Plexus Birth Injury than in unilateral Cerebral Palsy and suggests that both groups of children have affectively overcome their developmental disregard.IMPLICATIONS FOR REHABILITATIONChildren with Brachial Plexus Birth Injury frequently experience difficulties in activities of daily living.It has recently been suggested that children with Brachial Plexus Birth Injury may also show a non-use of the affected upper limb despite sufficient capacity, called developmental disregard.Children with Brachial Plexus Birth Injury and developmental disregard might therefore benefit from intensive therapies aimed at overcoming developmental disregard, originally developed for children with unilateral Cerebral Palsy.A combination of modified Constrained-Induced Movement Therapy with intensive Bimanual Training has shown to be affective in children with unilateral Cerebral Palsy.In a small sample, this study shows that a combination of modified Constrained-Induced Movement Therapy with intensive bimanual training is effective in children with Brachial Plexus Birth Injury, comparable and even more than in unilateral Cerebral Palsy.

Lateralized EEG mu power during action observation and motor imagery in typically developing children and children with unilateral Cerebral Palsy.

OBJECTIVE: During motor execution (ME), mu power is diminished over the contralateral hemisphere and increased over the ipsilateral hemisphere, which has been associated with cortical activation of the contralateral motor areas and inhibition of the ipsilateral motor areas respectively. The influence of action observation (AO) and motor imagery (MI) on mu power is less clear, especially in children, and remains to be studied in children with unilateral cerebral palsy (uCP). METHODS: We determined mu power during ME, AO, and MI of 45 typically developing (TD) children and 15 children with uCP over both hemispheres, for each hand. RESULTS: In TD children, over the left hemisphere mu power was lowered during ME when the right hand was used. In line, over the right hemisphere mu power was lowered when the left hand was addressed. In addition, during AO and MI increased mu power was observed when the right hand was addressed. In children with uCP, over the spared hemisphere mu power was diminished during ME when the less-affected hand was used. However, over the lesioned hemisphere, no mu changes were observed. CONCLUSIONS: The results of TD children fit the activation/inhibition model of mu power. SIGNIFICANCE: The results of children with uCP suggest that the lesioned hemisphere is unresponsive to the motor tasks.

Interventions to improve upper limb function for children with bilateral cerebral palsy: a systematic review.

AIM: To systematically review the efficacy of interventions on upper limb function in children 0 to 19 years of age with bilateral cerebral palsy on the basis of outcome measures of upper limb function and measures of activities and/or participation according to the International Classification of Functioning, Disability and Health. METHOD: Cochrane, PubMed, Embase, CINAHL, and Web of Science were searched from inception to September 2017. Methodological quality and strength of evidence were analysed by two independent raters using Sackett's level of evidence and the American Academy for Cerebral Palsy and Developmental Medicine (AACPDM) guidelines. RESULTS: Fifteen studies with a large variety of interventions and heterogeneity in outcome measures met the inclusion criteria. Twelve studies provided level IV evidence according to AACPDM guidelines. For three small randomized controlled trials the level of evidence was II. Only one of these trials showed strong methodological quality: a study on hand-arm bimanual intensive therapy including lower extremities. INTERPRETATION: We identified a large variety of interventions, heterogeneity in outcome measures, and generally weak to moderate methodological quality for most studies. We recommend further research specifically aimed at bimanual-intensive, goal-directed, and task-specific training programmes for the upper limb in children with bilateral cerebral palsy, using either high-quality (multicentre) trials or well-designed single-case trials. WHAT THIS PAPER ADDS: There is a large variety of interventions on upper limb function in children with bilateral cerebral palsy. Heterogeneity of outcome measures and interventions impeded firm conclusions about intervention efficacy. Most studies had low-level evidence and weak to moderate methodological quality. The strongest evidence from a small randomized controlled trial was for hand-arm bimanual intensive therapy including lower extremities.

Intervenciones para mejorar la función de las extremidades superiores en niños con parálisis cerebral bilateral: una revisión sistemática OBJETIVO: Revisar sistemáticamente la eficacia de la función de la extremidad superior con las intervenciones realizadas en niños de 0 a 19 años de edad con parálisis cerebral bilateral basada en medidas de la función de la extremidad superior, de actividades y / o participación, según la Clasificación Internacional del Funcionamiento, de la Discapacidad y de la Salud. MÉTODO: Se investigaron desde su inicio hasta septiembre del 2017 las siguientes bases de datos: Cochrane, PubMed, Embase, CINAHL y Web of Science. Tres evaluadores independientes analizaron la calidad metodológica y la calidad de la evidencia utilizando el nivel de evidencia de Sackett y las guias de la Academia Americana para la Parálisis Cerebral y Medicina del Desarrollo (AACPDM). RESULTADOS: Quince estudios con una gran variedad de intervenciones y heterogeneidad en las escalas de resultado cumplieron con los criterios de inclusión. Doce estudios proporcionaron evidencia nivel IV de acuerdo con las guías de la AACPDM. Otros tres ensayos pequeños controlados y aleatorios se clasificaron como nivel II de evidencia. Solo uno de estos ensayos mostró una calidad metodológica sólida que consista en un estudio sobre terapia bimanual intensiva mano-brazo que incluía extremidades inferiores. INTERPRETACIÓN: Se identificaron una gran variedad de intervenciones, heterogeneidad en las escalas de medición de los resultados, y en general una calidad metodológica de débil a moderada para la mayoría de los estudios. Recomendamos investigaciones adicionales dirigidas específicamente a programas de entrenamiento bimanual, orientado a objetivos específicos para la tarea del miembro superior en niños con parálisis cerebral bilateral, utilizando ensayos de alta calidad (multicéntricos) o ensayos dirigidos a estudiar solo un concepto bien diseñados.

Intervenções para melhorar a função do membro superior em crianças com paralisia cerebral bilateral: uma revisão sistemática OBJETIVO: Revisar sistematicamente a eficácia de intervenções para a função do membro superior em crianças de 0 a 19 anos de idade com paralisia cerebral bilateral com base em medidas de resultado da função do membro superior e medidas de atividades e/ou participação de acordo com a Classificação Internacional de Funcionalidade, Incapacidade e Saúde. MÉTODO: Cochrane, PubMed, Embase, CINAHL, e Web of Science foram pesquisadas do início até setembro de 2017. A qualidade metodológica e força da evidência foram analisados por três avaliadores independentes usando o nível Sackett's e evidência e as diretrizes da Academia Americana de Paralisia Cerebral e Medicina do Desenvolviemnto (AACPDM). RESULTADOS: Quinze estudos com uma grande variedade de intervenções e heterogeneidade de medidas de resultado atenderam aos critérios de inclusão. Doze estudos forneceram evidência nível IV de acordo com as diretrizes da AACPDM. Para três pequenos estudos randomizados controlados o nível de evidência foi II. Apenas um destes estudos mostrou forte qualidade metodológica: um estudo sobre terapia intensive bimanual mão-braço incluindo as extremidades inferiores. INTERPRETAÇÃO: Identificamos uma grande variedade de intervenções, heterogeneidade em medidas de resultado, e em geral qualidade metodológica de fraca a moderada para a maioria dos estudos. Recomendamos mais pesquisas especificamente voltadas para programas de treinamento intensivos bimanuais, direcionados a objetivos e específicos para tarefas para o membro superior de crianças com paralisia cerebral usando ou estudos de alta qualidade (multicêntricos) ou estudos de sujeito único bem desenhados.

Hand-Use-at-Home Questionnaire: validity and reliability in children with neonatal brachial plexus palsy or unilateral cerebral palsy.

OBJECTIVE: To investigate construct validity and test-retest reliability of the parent-rated Hand-Use-at-Home questionnaire (HUH) in children with neonatal brachial plexus palsy or unilateral cerebral palsy. DESIGN AND SUBJECTS: For this cross-sectional study, children with neonatal brachial plexus palsy or unilateral cerebral palsy, aged 3-10 years, were eligible. MAIN MEASURES: The HUH, Pediatric Outcome Data Collection Instrument Upper Extremity Scale (neonatal brachial plexus palsy only), and Children's Hand-Use Experience Questionnaire (unilateral cerebral palsy only) were completed. The HUH was completed twice in subgroups of both diagnoses. Lesion-extent (indication of involved nerve rootlets in neonatal brachial plexus palsy as confirmed during clinical observation and/or nerve surgery) and Manual Ability Classification System levels (unilateral cerebral palsy) were obtained from the medical records. Spearman correlation coefficients between the HUH and all clinical variables, agreement, standard error of measurement, smallest detectable change and intra-class correlation were calculated. RESULTS: A total of 260 patients participated (neonatal brachial plexus palsy: 181), of which 56 completed the second HUH (neonatal brachial plexus palsy: 16). Median age was 6.9 years for children with neonatal brachial plexus palsy, 116 had C5-C6 lesions. Median age for children with unilateral cerebral palsy was 6.4 years, 33 had Manual Ability Classification System Level II. The HUH correlated moderately with lesion-extent ( rs =-0.5), Pediatric Outcome Data Collection Instrument Upper Extremity Scale ( rs = 0.6) and Children's Hand-Use Experience Questionnaire ( rs = 0.5) but weakly with Manual Ability Classification System levels ( rs = -0.4). Test-retest reliability was excellent (intra-class correlation2,1 = 0.89, standard error of measurement = 0.599 and smallest detectable change = 1.66 logits) and agreement was good (mean difference HUH1 - HUH2 = 0.06 logits). CONCLUSION: The HUH showed good construct validity and test-retest reliability in children with neonatal brachial plexus palsy or unilateral cerebral palsy.

Process evaluation of two home-based bimanual training programs in children with unilateral cerebral palsy (the COAD-study): protocol for a mixed methods study.

BACKGROUND: As part of the COAD-study two home-based bimanual training programs for young children with unilateral Cerebral Palsy (uCP) have been developed, both consisting of a preparation phase and a home-based training phase. Parents are coached to use either an explicit or implicit motor learning approach while teaching bimanual activities to their child. A process evaluation of these complex interventions is crucial in order to draw accurate conclusions and provide recommendations for implementation in clinical practice and further research. The aim of the process evaluation is to systematically assess fidelity of the home-based training programs, to examine the mechanisms that contribute to their effects on child-related and parent-related outcomes, and to explore the influence of contextual factors. METHODS: A mixed methods embedded design is used that emerges from a pragmatism paradigm. The qualitative strand involves a generic qualitative approach. The process evaluation components fidelity (quality), dose delivered (completeness), dose received (exposure and satisfaction), recruitment and context will be investigated. Data collection includes registration of attendance of therapists and remedial educationalists to a course regarding the home-based training programs; a questionnaire to evaluate this course by the instructor; a report form concerning the preparation phase to be completed by the therapist; registration and video analyses of the home-based training; interviews with parents and questionnaires to be filled out by the therapist and remedial educationalist regarding the process of training; and focus groups with therapists and remedial educationalists as well as registration of drop-out rates and reasons, to evaluate the overall home-based training programs. Inductive thematic analysis will be used to analyse qualitative data. Qualitative and quantitative findings are merged through meta-inference. DISCUSSION: So far, effects of home-based training programs in paediatric rehabilitation have been studied without an extensive process evaluation. The findings of this process evaluation will have implications for clinical practice and further research regarding development and application of home-based bimanual training programs, executed by parents and aimed at improving activity performance and participation of children with uCP.

Home-based bimanual training based on motor learning principles in children with unilateral cerebral palsy and their parents (the COAD-study): rationale and protocols.

BACKGROUND: Home-based training is considered an important intervention in rehabilitation of children with unilateral cerebral palsy. Despite consensus on the value of home-based upper limb training, no evidence-based best practice exists. Promoting compliance of children to adhere to an intensive program while keeping parental stress levels low is an important challenge when designing home-based training programs. Incorporating implicit motor learning principles emerges to be a promising method to resolve this challenge. METHODS: Here we describe two protocols for home-based bimanual training programs, one based on implicit motor learning principles and one based on explicit motor learning principles, for children with unilateral spastic cerebral palsy aged 2 through 7 years. Children receive goal-oriented, task-specific bimanual training in their home environment from their parents for 3.5 h/week for 12 weeks according to an individualized program. Parents will be intensively coached by a multidisciplinary team, consisting of a pediatric therapist and remedial educationalist. Both programs consist of a preparation phase (goal setting, introductory meetings with coaching professionals, design of individualized program, instruction of parents, home visit) and home-based training phase (training, video-recordings, registrations, and telecoaching and home visits by the coaching team). The programs contrast with respect to the teaching strategy, i.e. how the parents support their child during training. In both programs parents provide their child with instructions and feedback that focus on the activity (i.e. task-oriented) or the result of the activity (i.e. result-oriented). However, in the explicit program parents are in addition instructed to give exact instructions and feedback on the motor performance of the bimanual activities, whereas in the implicit program the use of both hands and the appropriate motor performance of the activity are elicited via manipulation of the organization of the activities. DISCUSSION: With the protocols described here, we aim to take a next step in the development of much needed evidence-based home-based training programs for children with unilateral cerebral palsy.

Development and psychometric properties of the Hand-Use-at-Home questionnaire to assess amount of affected hand-use in children with unilateral paresis.

AIM: To describe the development of the parent-rated Hand-Use-at-Home questionnaire (HUH) assessing the amount of spontaneous use of the affected hand in children with unilateral paresis, and to test its internal structure, unidimensionality, and validity. METHOD: Parents of children with unilateral cerebral palsy (CP) and professionals participated in the development of the HUH. To examine internal validity, data of 322 children (158 males, 164 females; mean age 6y 7mo, standard deviation [SD] 2y 1mo) with unilateral CP (n=131) or neonatal brachial plexus palsy (NBPP) (n=191) were collected. Rasch analysis was used to examine discriminative capacity of the 5-category rating scale as well as unidimensionality and hierarchy of the item set. Additionally, data of 55 children with typical development (24 males, 31 females; 6y 9mo, SD 2y 5mo) were used to examine construct validity. RESULTS: The 5-category rating scale was disordered in all items and was collapsed to obtain the best discriminating sum score. Ten misfitting or redundant items were removed. Eighteen hierarchically ordered bimanual items fitted the unidimensional model within acceptable range. The HUH significantly discriminated between the three groups (children with typical development, NBPP, unilateral CP; H(2) =118.985, p<0.001), supporting its construct validity. INTERPRETATION: The HUH is a valid instrument to assess the amount of spontaneous use of the affected hand in children with unilateral upper-limb paresis.

The relation between mirror movements and non-use of the affected hand in children with unilateral cerebral palsy.

AIM: In children with unilateral cerebral palsy (CP), it is widely believed that mirror movements contribute to non-use of the affected hand despite preserved capacity, a phenomenon referred to as developmental disregard. We aimed to test whether mirror movements are related to developmental disregard, and to clarify the relation between mirror movements and bimanual function. METHOD: A repetitive squeezing task simultaneously measuring both hands' grip-forces was developed to assess mirror movements by using maximum cross-correlation coefficient (CCCmax ) as well as strength measures (MMstrength ). Developmental disregard, bimanual performance, and capacity were assessed using a validated video-observation method. Twenty-one children with unilateral CP participated (Median age 10y 7mo, interquartile range [IQR] 10y 1mo-12y 9mo). Outcome measures of mirror movements were correlated to developmental disregard, bimanual performance, and capacity scores using Spearman's correlations (significance level: α<0.05). RESULTS: Mirror movements were not related to developmental disregard. However, enhanced mirror movements in the less-affected hand were related to reduced performance (CCCmax : ρ=-0.526, p=0.007; MMstrength : ρ=-0.750, p<0.001) and capacity (CCCmax : ρ=-0.410, p=0.033; MMstrength : ρ=-0.679, p<0.001). These relations were only moderate (performance:MMstrength : ρ=-0.504, p=0.010), low (capacity: MMstrength : ρ=-0.470, p=0.016) or absent for mirror movements in the affected hand. Additionally, seven children showed stronger movements in their less-affected hands when actually being asked to move their affected hand. INTERPRETATION: These findings show no relation between mirror movements and developmental disregard, but support an association between mirror movements and bimanual function.

Event-related Potentials During Target-response Tasks to Study Cognitive Processes of Upper Limb Use in Children with Unilateral Cerebral Palsy.

Unilateral Cerebral Palsy (CP) is a neurodevelopmental disorder that is a very common cause of disability in childhood. It is characterized by unilateral motor impairments that are frequently dominated in the upper limb. In addition to a reduced movement capacity of the affected upper limb, several children with unilateral CP show a reduced awareness of the remaining movement capacity of that limb. This phenomenon of disregarding the preserved capacity of the affected upper limb is regularly referred to as Developmental Disregard (DD). Different theories have been postulated to explain DD, each suggesting slightly different guidelines for therapy. Still, cognitive processes that might additionally contribute to DD in children with unilateral CP have never been directly studied. The current protocol was developed to study cognitive aspects involved in upper limb control in children with unilateral CP with and without DD. This was done by recording event-related potentials (ERPs) extracted from the ongoing EEG during target-response tasks asking for a hand-movement response. ERPs consist of several components, each of them associated with a well-defined cognitive process (e.g., the N1 with early attention processes, the N2 with cognitive control and the P3 with cognitive load and mental effort). Due to its excellent temporal resolution, the ERP technique enables to study several covert cognitive processes preceding overt motor responses and thus allows insight into the cognitive processes that might contribute to the phenomenon of DD. Using this protocol adds a new level of explanation to existing behavioral studies and opens new avenues to the broader implementation of research on cognitive aspects of developmental movement restrictions in children.

Kinematic parameters of hand movement during a disparate bimanual movement task in children with unilateral Cerebral Palsy.

Children with unilateral Cerebral Palsy (uCP) experience problems performing tasks requiring the coordinated use of both hands (bimanual coordination; BC). Additionally, some children with uCP display involuntary symmetrical activation of the opposing hand (mirrored movements). Measures, used to investigate therapy-related improvements focus on the functionality of the affected hand during unimanual or bimanual tasks. None however specifically address spatiotemporal integration of both hands. We explored the kinematics of hand movements during a bimanual task to identify parameters of BC. Thirty-seven children (aged 10.9±2.6years, 20 male) diagnosed with uCP participated. 3D kinematic motion analysis was performed during the task requiring opening of a box with their affected- (AH) or less-affected hand (LAH), and pressing a button inside with the opposite hand. Temporal and spatial components of data were extracted and related to measures of hand function and level of impairment. Total task duration was correlated with the Jebsen-Taylor Test of Hand Function in both conditions (either hand leading with the lid-opening). Spatial accuracy of the LAH when the box was opened with their AH was correlated with outcomes on the Children's Hand Use Experience Questionnaire. Additionally, we found a subgroup of children displaying non-symmetrical movement interference associated with greater movement overlap when their affected hand opened the box. This subgroup also demonstrated decreased use of the affected hand during bimanual tasks. Further investigation of bimanual interference, which goes beyond small scaled symmetrical mirrored movements, is needed to consider its impact on bimanual task performance following early unilateral brain injury.

Children with unilateral cerebral palsy show diminished implicit motor imagery with the affected hand.

AIM: Motor imagery refers to the mental simulation of a motor action without producing an overt movement. Implicit motor imagery can be regarded as a first-person kinesthetic perceptual judgement, and addresses the capacity to engage into the manipulation of one's body schema. In this study, we examined whether children with unilateral cerebral palsy (CP) are able to engage in implicit motor imagery. METHOD: A modified version of the hand laterality judgment task was employed. Erroneous responses, reaction times, and event-related potentials from the electroencephalograph were analysed. RESULTS: In 13 children with typical development (mean age 10y 7mo, SD 1y 2mo; seven male, six female), we observed the classic rotation direction effect. Specifically, when comparing outward rotated with inward rotated hand pictures, decreased accuracy and increased response times were observed. Event-related potentials analyses of the electroencephalogram revealed a more marked N1 and an enhanced rotation-related negativity. INTERPRETATION: These findings suggest that an implicit motor imagery strategy was used to solve the task. However, in 10 children with unilateral CP (mean age 10y 7mo, SD 2y 5mo; five male, five female), these effects were observed only when the less-affected hand was involved. This observation suggests that children with CP could benefit from visual training strategies.

Intensive upper limb intervention with self-management training is feasible and promising for older children and adolescents with unilateral cerebral palsy.

This study explored the feasibility and preliminary effectiveness of a short (one week) intensive intervention combining Constraint Induced Movement Therapy (CIMT) and bimanual training (BiT) to improve upper limb capacity and bimanual performance guided by individual goal setting in children and adolescents with unilateral cerebral palsy aged 8-18 years. Self-management training was added to the intervention to maximize the effect of training and to empower the participants in self-monitoring the effective use of their affected hand. Functional goals (Canadian Occupational Performance Measure), unimanual capacity (Box and Block Test), bimanual performance (ABILHAND-Kids, Children's Hand-use Experience Questionnaire (CHEQ)) and amount of use (Video Observation Aarts and Aarts - determine developmental disregard (VOAA-DDD-R)) were measured at baseline, one week and four months post intervention. Twenty children (mean age 9.5 years) participated. Repeated measures ANOVA was used to measure effects over time. Compared to baseline, there were significant improvements on all outcome measures. The largest effect sizes were found for the COPM-performance and COPM-satisfaction (Cohen's d=2.09 and d=2.42, respectively). The effect size was large for the ABILHAND-Kids (d=0.86), moderate for the CHEQ (d=0.70) and Box and Block Test (d=0.56), and small for the VOAA-DDD-R (d=0.33). All effects were retained at the four months post intervention assessment. The results of this study indicate that one-week (36h) intensive CIMT-BiT combined with self-management training is a feasible and promising intervention for improving the capacity of the upper limb and its use in bimanual activities in older children and adolescents with unilateral CP.

Neglect-like characteristics of developmental disregard in children with cerebral palsy revealed by event related potentials.

BACKGROUND: Children with unilateral Cerebral Palsy (CP) often show diminished awareness of the remaining capacity of their affected upper limb. This phenomenon is known as Developmental Disregard (DD). DD has been explained by operant conditioning. Alternatively, DD can be described as a developmental delay resulting from a lack of use of the affected hand during crucial developmental periods. We hypothesize that this delay is associated with a general delay in executive functions (EF) related to motor behavior, also known as motor EFs. METHODS: Twenty-four children with unilateral CP participated in this cross-sectional study, twelve of them diagnosed with DD. To test motor EFs, a modified go/nogo task was presented in which cues followed by go- or nogo-stimuli appeared at either the left or right side of a screen. Children had to press a button with the hand corresponding to the side of stimulus presentation. Apart from response accuracy, Event-Related Potentials (ERPs) extracted from the ongoing EEG were used to register covert cognitive processes. ERP N1, P2, N2, and P3 components elicited by cue-, go-, and nogo-stimuli were further analyzed to differentiate between different covert cognitive processes. RESULTS: Children with DD made more errors. With respect to the ERPs, the P3 component to go-stimuli was enhanced in children with DD. This enhancement was related to age, such that younger children with DD showed stronger enhancements. In addition, in DD the N1 component to cue- and go-stimuli was decreased. CONCLUSIONS: The behavioral results show that children with DD experience difficulties when performing the task. The finding of an enhanced P3 component to go-stimuli suggests that these difficulties are due to increased mental effort preceding movement. As age in DD mediated this enhancement, it seems that this increased mental effort is related to a developmental delay. The additional finding of a decreased N1 component in DD furthermore suggests a general diminished visuo-spatial attention. This effect reveals that DD might be a neuropsychological phenomenon similar to post-stroke neglect syndrome that does not resolve during development. These findings suggest that therapies aimed at reducing neglect could be a promising addition to existing therapies for DD.

Assessment of upper limb capacity in children with unilateral cerebral palsy: construct validity of a Rasch-reduced Modified House Classification.

AIM: The aim of this study was to test and improve the unidimensionality and item hierarchy of the Modified House Classification (MHC) for the assessment of upper limb capacity in children with unilateral cerebral palsy (CP) using Rasch analysis. The construct validity of the Rasch-reduced item set was evaluated. METHOD: Modified House Classification items were scored from 369 videotaped assessments of 159 children with unilateral CP (98 males, 61 females; median age 6y 6mo, range 2y 1mo-17y 5mo). Construct validity was tested in 40 other children with unilateral CP (21 males, 19 females; median age 8y 2mo, range 3y 3mo-17y 6mo) by comparing total scores with the Manual Ability Classification System (MACS) and the ABILHAND-Kids scale. RESULTS: Fifteen MHC items could be included in the Rasch analysis. The excluded items were either too easy or too difficult. Fourteen items fitted the unidimensional model (χ(2) =41.3, df=39, p=0.37). The hierarchy of these items was different from the original MHC. There was a significant correlation with the MACS (r=-0.901, p<0.001) and the ABILHAND-Kids scale (r=0.558, p<0.001). INTERPRETATION: The original item hierarchy of the MHC can be improved in order to use its sum score for the assessment of upper limb capacity in children with unilateral CP. The Rasch-reduced 14-item MHC with weighted sum score shows good construct validity to measure functional capacity of the affected hand in children with unilateral CP.

Unravelling developmental disregard in children with unilateral cerebral palsy by measuring event-related potentials during a simple and complex task.

BACKGROUND: In a subset of children with unilateral Cerebral Palsy (CP) a discrepancy between capacity and performance of the affected upper limb can be observed. This discrepancy is known as Developmental Disregard (DD). Though the phenomenon of DD has been well documented, its underlying cause is still under debate. DD has originally been explained based on principles of operant conditioning. Alternatively, it has been proposed that DD results from a diminished automaticity of movements, resulting in an increased cognitive load when using the affected hand. To investigate the amount of involved cognitive load we studied Event-Related Potentials (ERPs) preceding task-related motor responses during a single-hand capacity and a dual-hand performance task. It was hypothesised that children with DD show alterations related to long-latency ERP components when selecting a response with the affected upper limb, reflecting increased cognitive load in order to generate an adequate response and especially so within the dual-hand task. METHODS: Fifteen children with unilateral CP participated in the study. One of the participants was excluded due to major visual impairments. Seven of the remaining participants displayed DD. The other seven children served as a control group. All participants performed two versions of a cue-target paradigm, a single-hand capacity and a dual-hand performance task. The ERP components linked to target presentation were inspected: the mid-latency P2 component and the consecutive long-latency N2b component. RESULTS: In the dual-hand performance task children with DD showed an enhancement in mean amplitude of the long-latency N2b component when selecting a response with their affected hand. No differences were found regarding the amplitude of the mid-latency P2 component. No differences were observed regarding the single-hand capacity task. The control group did not display any differences in ERPs linked to target evaluation processes between both hands. CONCLUSION: These electrophysiological findings show that DD is associated with increased cognitive load when movements are prepared with the affected hand during a dual-hand performance task. These findings confirm behavioural observations, advance our insights on the neural substrate of DD and have implications for therapy.

Guidelines for future research in constraint-induced movement therapy for children with unilateral cerebral palsy: an expert consensus.

AIM: The aim of this study was to provide an overview of what is known about constraintinduced movement therapy (CIMT) in children with unilateral cerebral palsy (CP), to identify current knowledge gaps, and to provide suggestions for future research. METHOD: Nine experts participated in a consensus meeting. A comprehensive literature search was conducted and data were summarized before the meeting. The core model produced by the European network for Health Technology Assessment was used as a framework for discussion and to identify critical issues for future research. RESULTS: All models of CIMT have demonstrated improvements in the upper limb abilities of children with unilateral CP. A consensus was reached on 11 important questions to be further explored in future studies. The areas of highest priority included the effect of dosage, the effect of repeated CIMT, and the impact of predictive factors, such as age, on the response to CIMT. Consensus suggestions for future study designs and the use of validated outcome measures were also provided. INTERPRETATION: The CIMT construct is complex, and much remains unknown. It is unclear whether a specific model of CIMT demonstrates superiority over others and whether dosage of training matters. Future research should build upon existing knowledge and aim to provide information that will help implement CIMT in various countries with different healthcare resources and organizational structures.