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).

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.

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.

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.

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.

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.

Assessment of upper-limb capacity, performance, and developmental disregard in children with cerebral palsy: validity and reliability of the revised Video-Observation Aarts and Aarts module: Determine Developmental Disregard (VOAA-DDD-R).

AIM: To investigate the validity and reliability of the revised Video-Observation Aarts and Aarts module: Determine Developmental Disregard (VOAA-DDD-R). METHOD: Upper-limb capacity and performance were assessed in children with unilateral spastic cerebral palsy (CP) by measuring overall duration of affected upper-limb use and the frequency of specific behaviours during a task in which bimanual activity was demanded ('stringing beads') and stimulated ('decorating a muffin'). Developmental disregard was defined as the difference in duration of affected upper-limb use between both tasks. Raters were two occupational and one physical therapist who received 3 hours of training. Construct validity was determined by comparing children with CP with typically developing children. Intrarater, interrater, and test-retest reliability were determined using the intraclass correlation coefficient. Standard errors of measurement and smallest detectable differences were also calculated. RESULTS: Twenty-five children with CP (15 females, 10 males; mean age 4 y 9 mo [SD 1 y 7 mo], range 2 y 9 mo-8 y; Manual Ability Classification System levels I-III) scored lower on capacity (p=0.052) and performance (p<0.001), and higher on developmental disregard (p<0.001) than 46 age- and sex-matched typically developing children (23 males; mean age 5 y 3 mo [SD 1 y 5 mo], range 2 y 6 mo-8 y). The intraclass correlation coefficients (0.79-1.00) indicated good reliability. Absolute agreement was high, standard errors of measurement ranged from 4.5 to 6.8%, and smallest detectable differences ranged from 12.5 to 19.0%. INTERPRETATION: The VOAA-DDD-R can be reliably and validly used by occupational and physical therapists to assess upper-limb capacity, performance, and developmental disregard in children (2 y 6 mo-8 y) with CP.

A neurocognitive perspective on developmental disregard in children with hemiplegic cerebral palsy.

A common problem in children with hemiplegic cerebral palsy (CP) is the asymmetrical development of arm and hand capacity caused by the lack of use of the affected upper limb, or developmental disregard. In this paper, we provide a neuropsychological model that relates developmental disregard to attentional processes and motor learning. From this model, we hypothesize that high attentional demands associated with the use of the affected upper limb might hinder its use in daily life, and therefore may be a factor in developmental disregard. This can be assessed with a dual-task paradigm. However, until now, this has not been applied to children with CP. We provide recommendations for using a dual-task paradigm in children with CP based on empirical studies in typically developing children and children with developmental coordination disorder. Ultimately, these dual-task studies may be used to improve interventions aimed at reducing developmental disregard.

Standardized tests of handwriting readiness: a systematic review of the literature.

AIM: To establish if there are psychometrically sound standardized tests or test items to assess handwriting readiness in 5- and 6-year-old children on the levels of occupations activities/tasks and performance. METHOD: Electronic databases were searched to identify measurement instruments. Tests were included in a systematic review if: (1) participants are 5 and 6 years old, (2) the focus was on handwriting readiness, and (3) the measurement was standardized. In the second step a further electronic search was undertaken for selected relevant measurement instruments to evaluate the content, psychometric properties, and feasibility of these instruments. RESULTS: The search identified 1113 citations. In the final selection 39 articles with information about 12 tests were included. The content, feasibility, and psychometric properties of these 12 tests were evaluated and none of the instruments was satisfactory, according to the specific criteria. INTERPRETATION: None of the instruments include all necessary components to evaluate writing readiness. Therefore, the development of an all encompassing assessment is necessary to test handwriting readiness and to make tailored interventions possible.

Validity and reliability of the VOAA-DDD to assess spontaneous hand use with a video observation tool in children with spastic unilateral cerebral palsy.

BACKGROUND: In 2003 new computer software, the VOAA (Video Observations Aarts and Aarts), was designed to score and evaluate two important aspects of spontaneous upper limb use, i.e. overall duration and frequency of specific behaviours. The aim of this study was to investigate the test-retest, interrater and intrarater reliability and the construct validity of a new module, the VOAA-DDD, to determine developmental disregard in children with spastic unilateral cerebral palsy (CP). METHODS: A test-retest design with three raters for reliability and a two-group design for construct validity were used. Subjects were a total of 20 children with spastic unilateral CP equally divided in two age groups (2.5-5 and 5-8 years), and 56 healthy children of the same age groups. Overall duration and frequency of specific behaviours of the affected arm and hand were assessed during a task demanding ('stringing beads') and a task stimulating ('decorating a muffin') the use of both hands. Reliability was estimated by intraclass correlation coefficients (ICCs). Construct validity was assessed by comparing children with CP to healthy children. RESULTS: All ICCs exceeded 0.87. In contrast with healthy children, children with CP used their affected hand less during the 'muffin' task compared to the 'beads' task. Of the children with CP, 90% in the age group of 2.5-5 years and 50% in the age group of 5-8 years showed values exceeding the extreme values of healthy controls, respectively, indicating developmental disregard. CONCLUSION: The VOAA-DDD is a reliable and valid instrument to assess spontaneous use of the affected arm and hand in order to determine developmental disregard in children with spastic unilateral CP.