Effect of Home-based Bimanual Training in Children with Unilateral Cerebral Palsy (The COAD-study): A Case Series.

Purpose: To explore the child- and parent-related effects of home-based bimanual training in children with unilateral cerebral palsy.Methods: Case series of 14 children (2-7 years) who completed goal-oriented task-specific training for 3.5 hours/week for 12 weeks by a program adopting implicit (n = 5) or explicit (n = 9) motor learning. A therapist and remedial educationalist coached parents. Progression on bimanual goals (Canadian Occupational Performance Measure (COPM)) and therapy-related parental stress (interviews) were of primary interest. Data were collected at baseline (T0), halfway through and at the end of training (T1 and T2), and after 12 weeks (T3).Results: On the COPM performance scale a clinically relevant change was seen in 50% (7/14), 86% (12/14), and 85% (11/13) of the children, at T1, T2, and T3, respectively. Some parents indicated that they had experienced stress because of the training intensity.Conclusion: The child- and parent-related effects of the home-based bimanual training programs are encouraging.

Explicit and implicit motor sequence learning in children and adults; the role of age and visual working memory.

This study investigated explicit and implicit motor learning, and the influence of visual working memory (VWM) and age. Sixty children and 28 adults learned a nine-button sequence task explicitly and implicitly. Performance in explicit and implicit learning improved with age. Learning curves were similar across ages for implicit learning. In explicit learning, learning curves differed across ages: younger children started slower, but their learning rate was higher compared to older children. Learning curves were similar across VWM scores, but performance in explicit learning was positively influenced by VWM scores. Further research and implications for education and rehabilitation are discussed.

The validity and reliability of the Functional Strength Measurement (FSM) in children with intellectual disabilities.

BACKGROUND: There is lack of valid and reliable field-based tests for assessing functional strength in young children with mild intellectual disabilities (IDs). AIM: The aim of this study was to investigate the test-retest reliability and construct validity of the Functional Strength Measurement in children with ID (FSM-ID). METHOD: Fifty-two children with mild ID (40 boys and 12 girls, mean age 8.48 years, SD = 1.48) were tested with the FSM. Test-retest reliability (n = 32) was examined by a two-way interclass correlation coefficient for agreement (ICC 2.1A). Standard error of measurement and smallest detectable change were calculated. Construct validity was determined by calculating correlations between the FSM-ID and handheld dynamometry (HHD) (convergent validity), FSM-ID, FSM-ID and subtest strength of the Bruininks-Oseretsky test of motor proficiency - second edition (BOT-2) (convergent validity) and the FSM-ID and balance subtest of the BOT-2 (discriminant validity). RESULTS: Test-retest reliability ICC ranged 0.89-0.98. Correlation between the items of the FSM-ID and HHD ranged 0.39-0.79 and between FSM-ID and BOT-2 (strength items) 0.41-0.80. Correlation between items of the FSM-ID and BOT-2 (balance items) ranged 0.41-0.70. CONCLUSION: The FSM-ID showed good test-retest reliability and good convergent validity with the HHD and BOT-2 subtest strength. The correlations assessing discriminant validity were higher than expected. Poor levels of postural control and core stability in children with mild IDs may be the underlying factor of those higher correlations.

Learning New Letter-like Writing Patterns Explicitly and Implicitly in Children and Adults.

A handwriting task was used to test the assumption that explicit learning is dependent on age and working memory, while implicit learning is not. The effect of age was examined by testing both, typically developing children (5-12 years old, n = 81) and adults (n = 27) in a counterbalanced within-subjects design. Participants were asked to repeatedly write letter-like patterns on a digitizer with a non-inking pen. Reproduction of the pattern was better after explicit learning compared to implicit learning. Age had positive effects on both explicit and implicit learning; working memory did not affect learning in either conditions. These results show that it may be more effective to learn writing new letter-like patterns explicitly and that an explicit teaching method is preferred in mainstream primary education.

Feasibility and effect of home-based therapy programmes for children with cerebral palsy: a protocol for a systematic review.

INTRODUCTION: Given the promising advantages of upper extremity home-based programmes in children with cerebral palsy (CP), a systematic review of the available literature on this topic is warranted. The purpose of the systematic review described in this protocol is to investigate currently available home-based occupational therapy and physiotherapy programmes regarding both their feasibility and effect. METHODS AND ANALYSIS: This protocol describes a systematic review, developed in accordance with the Preferred Reporting Items for Systematic Review and Meta-Analysis Protocols (PRISMA-P) 2015. Studies will be included in which primary data are collected, participants are children aged <18 years with any type of CP and the intervention of interest is a home-based occupational therapy or physiotherapy intervention. Comparators of interest are: no therapy, care as usual, centre-based occupational therapy or physiotherapy, an alternative home-based programme and a medical intervention. Studies will be included that report either on feasibility (ie, acceptability, demand, implementation, practicality, adaptation, expansion or integration) or on efficacy/effectiveness (ie, child-related upper extremity outcomes within all International Classification of Functioning, Disability and Health levels or parent-related/caregiver-related outcomes on the psychological and social domain). Relevant studies will be identified by searching the databases MEDLINE, EMBASE, CINAHL, PsycINFO, PEDro, OTSeeker and CPCI-S as well as the trial registers ICTRP and CENTRAL, the reference lists of included records and by circulating a bibliography of the included records to authors of included studies. There will be no restrictions on language or year of publication. The search strategy consists of terms related to the population and intervention. Data will be extracted in duplicate using a digital data extraction form. ETHICS AND DISSEMINATION: The proposed study does not involve collection of primary data. Accordingly, no ethical approval is required. The authors will disseminate the findings of this systematic review through publication in a peer-reviewed journal and conference presentation(s). TRIAL REGISTRATION NUMBER: CRD42016043743; pre-results.

Development and face validity of a cerebral visual impairment motor questionnaire for children with cerebral palsy.

AIM: The objectives of this study were (i) to develop two cerebral visual impairment motor questionnaires (CVI-MQ's) for children with cerebral palsy (CP): one for children with Gross Motor Function Classification System (GMFCS) levels I, II and III and one for children with GMFCS levels IV and V; (ii) to describe their face validity and usability; and (iii) to determine their sensitivity and specificity. BACKGROUNDS: The initial versions of the two CVI-MQ's were developed based on literature. Subsequently, the Delphi method was used in two groups of experts, one familiar with CVI and one not familiar with CVI, in order to gain consensus about face validity and usability. The sensitivity and specificity of the CVI-MQ's were subsequently assessed in 82 children with CP with (n = 39) and without CVI (n = 43). With the receiver operating curve the cut-off scores were determined to detect possible presence or absence of CVI in children with CP. RESULTS: Both questionnaires showed very good face validity (percentage agreement above 96%) and good usability (percentage agreement 95%) for practical use. The CVI-MQ version for GMFCS levels I, II and III had a sensitivity of 1.00 and specificity of 0.96, with a cut-off score of 12 points or higher, and the version for GMFCS levels IV and V had a sensitivity of 0.97 and a specificity of 0.98, with a cut-off score of eight points or higher. CONCLUSION: The CVI-MQ is able to identify at-risk children with CP for the probability of having CVI.

Gross motor function in children with spastic Cerebral Palsy and Cerebral Visual Impairment: A comparison between outcomes of the original and the Cerebral Visual Impairment adapted Gross Motor Function Measure-88 (GMFM-88-CVI).

PURPOSE: To investigate whether the adapted version of the Gross Motor Function Measure-88 (GMFM-88) for children with Cerebral Palsy (CP) and Cerebral Visual Impairment (CVI) results in higher scores. This is most likely to be a reflection of their gross motor function, however it may be the result of a better comprehension of the instruction of the adapted version. METHOD: The scores of the original and adapted GMFM-88 were compared in the same group of children (n=21 boys and n=16 girls), mean (SD) age 113 (30) months with CP and CVI, within a time span of two weeks. A paediatric physical therapist familiar with the child assessed both tests in random order. The GMFCS level, mental development and age at testing were also collected. The Wilcoxon signed-rank test was used to compare two different measurements (the original and adapted GMFM-88) on a single sample, (the same child with CP and CVI; p<0.05). RESULTS: The comparison between scores on the original and adapted GMFM-88 in all children with CP and CVI showed a positive difference in percentage score on at least one of the five dimensions and positive percentage scores for the two versions differed on all five dimensions for fourteen children. For six children a difference was seen in four dimensions and in 10 children difference was present in three dimensions (GMFM dimension A, B& C or C, D & E) (p<0.001). CONCLUSION: The adapted GMFM-88 provides a better estimate of gross motor function per se in children with CP and CVI that is not adversely impacted bytheir visual problems. On the basis of these findings, we recommend using the adapted GMFM-88 to measure gross motor functioning in children with CP and CVI.

Learning to echolocate in sighted people: a correlational study on attention, working memory and spatial abilities.

Echolocation can be beneficial for the orientation and mobility of visually impaired people. Research has shown considerable individual differences for acquiring this skill. However, individual characteristics that affect the learning of echolocation are largely unknown. In the present study, we examined individual factors that are likely to affect learning to echolocate: sustained and divided attention, working memory, and spatial abilities. To that aim, sighted participants with normal hearing performed an echolocation task that was adapted from a previously reported size-discrimination task. In line with existing studies, we found large individual differences in echolocation ability. We also found indications that participants were able to improve their echolocation ability. Furthermore, we found a significant positive correlation between improvement in echolocation and sustained and divided attention, as measured in the PASAT. No significant correlations were found with our tests regarding working memory and spatial abilities. These findings may have implications for the development of guidelines for training echolocation that are tailored to the individual with a visual impairment.

Revealing hot executive function in children with motor coordination problems: What's the go?

Recent research suggests that children with Developmental Coordination Disorder (DCD) often show deficits in executive functioning (EF) and, more specifically, the ability to use inhibitory control in 'hot', emotionally rewarding contexts. This study optimized the assessment of sensitivity of children with DCD to emotionally significant stimuli by using easily discriminable emotional expressions in a go/no-go task. Thirty-six children (12 with DCD), aged 7-12years, completed an emotional go/no-go task in which neutral facial expressions were paired with either happy or sad ones. Each expression was used as both, a go and no-go target in different runs of the task. There were no group differences in omission errors; however, the DCD group made significantly more commission errors to happy no-go faces. The particular pattern of performance in DCD confirms earlier reports of (hot) EF deficits. Specifically, a problem of inhibitory control appears to underlie the atypical pattern of performance seen in DCD on both cold and hot EF tasks. Disrupted coupling between cognitive control and emotion processing networks, such as fronto-parietal and fronto-striatal networks, may contribute to reduced inhibitory control in DCD. The implications for a broader theoretical account of DCD are discussed, as are implications for intervention.

Reliability of the modified Gross Motor Function Measure-88 (GMFM-88) for children with both Spastic Cerebral Palsy and Cerebral Visual Impairment: A preliminary study.

PURPOSE: The aims of this study were to adapt the Gross Motor Function Measure-88 (GMFM-88) for children with Cerebral Palsy (CP) and Cerebral Visual Impairment (CVI) and to determine the test-retest and interobserver reliability of the adapted version. METHOD: Sixteen paediatric physical therapists familiar with CVI participated in the adaptation process. The Delphi method was used to gain consensus among a panel of experts. Seventy-seven children with CP and CVI (44 boys and 33 girls, aged between 50 and 144 months) participated in this study. To assess test-retest and interobserver reliability, the GMFM-88 was administered twice within three weeks (Mean=9 days, SD=6 days) by trained paediatric physical therapists, one of whom was familiar with the child and one who wasn't. Percentages of identical scores, Cronbach's alphas and intraclass correlation coefficients (ICC) were computed for each dimension level. RESULTS: All experts agreed on the proposed adaptations of the GMFM-88 for children with CP and CVI. Test-retest reliability ICCs for dimension scores were between 0.94 and 1.00, mean percentages of identical scores between 29 and 71, and interobserver reliability ICCs of the adapted GMFM-88 were 0.99-1.00 for dimension scores. Mean percentages of identical scores varied between 53 and 91. Test-retest and interobserver reliability of the GMFM-88-CVI for children with CP and CVI was excellent. Internal consistency of dimension scores lay between 0.97 and 1.00. CONCLUSION: The psychometric properties of the adapted GMFM-88 for children with CP and CVI are reliable and comparable to the original GMFM-88.

From numeracy to arithmetic: Precursors of arithmetic performance in children with cerebral palsy from 6 till 8 years of age.

Children with cerebral palsy (CP) are generally delayed in arithmetic compared to their peers. The development of early numeracy performance in children with CP is not yet evident, nor have the factors associated with change over time been identified. Therefore, we examined the development of numeracy in children with CP over a two year period and studied which cognitive factors were predictive of arithmetic performance. A longitudinal study with three measurement waves separated by one year was conducted. 56 children participated (37 boys, M=6.0 years, SD=.58). Standardized tasks were used to assess verbal- and visual-spatial working memory, executive functioning, fine motor skills and early numeracy performance. In addition, experimental tasks were developed to measure counting and arithmetic. The results showed that early numeracy performance of children with CP increased between 6 and 8 years of age. Structural equation modelling showed that early numeracy was strongly related to arithmetic performance at the consecutive year. Working memory, counting and fine motor skills were all positively related to early numeracy performance a year later. Furthermore, working memory and fine motor skills were precursors of the development of early numeracy. Considering the importance of numeracy and arithmetic in daily life and in academic and work success, children with CP could substantially benefit from intervention programs aimed at increasing working memory and early numeracy performance.

Reliability of the modified Paediatric Evaluation of Disability Inventory, Dutch version (PEDI-NL) for children with cerebral palsy and cerebral visual impairment.

PURPOSE: The aims of this study were to adapt the Paediatric Evaluation of Disability Inventory, Dutch version (PEDI-NL) for children with cerebral visual impairment (CVI) and cerebral palsy (CP) and determine test-retest and inter-respondent reliability. METHOD: The Delphi method was used to gain consensus among twenty-one health experts familiar with CVI. Test-retest and inter-respondent reliability were assessed for parents and caregivers of 75 children (aged 50-144 months) with CP and CVI. The percentage identical scores of item scores were computed, as well as the interclass coefficients (ICC) and Cronbach's alphas of scale scores over the domains self-care, mobility, and social function. RESULTS: All experts agreed on the adaptation of the PEDI-NL for children with CVI. On item score, for the Functional Skills scale, mean percentage identical scores variations for test-retest reliability were 73-79 with Caregiver Assistance scale 73-81, and for inter-respondent reliability 21-76 with Caregiver Assistance scale 40-43. For all scales over all domains ICCs exceeded 0.87. For the domains self-care, mobility, and social function, the Functional Skills scale and the Caregiver Assistance scale have Cronbach's alpha above 0.88. CONCLUSION: The adapted PEDI-NL for children with CP and CVI is reliable and comparable to the original PEDI-NL.

Deficits of hot executive function in developmental coordination disorder: sensitivity to positive social cues.

Recent research shows that children with motor coordination problems (or developmental coordination disorder - DCD) show deficits in not only cool executive function (EF), but also hot EF. We aimed to determine whether this deficit of hot EF is due to heightened sensitivity to rewarding stimuli, specifically, or to a general deficit of cognitive control, like inhibition. Using two versions of a go/no-go task, one with neutral facial expressions and the other with happy and fearful faces, we compared 12 children with DCD with 28 typically-developing children, aged 7-12 years. Like earlier studies, children responded faster to happy faces. Both groups showed comparable accuracy in response to go targets, and also had similar commission errors, except when the no-go stimulus was a happy face. Importantly, the DCD group made significantly more commission errors to happy faces failing to suppress their response on more than half of the no-go trials. These results suggest a heightened sensitivity to emotionally significant distractors in DCD; this type of impulsivity may undermine self-regulation in DCD, with possible implications for adaptive function and emotional well-being. We argue that the interaction of cognitive control and emotion processing networks may be disrupted in DCD or delayed in development.

Cognitive precursors of arithmetic development in primary school children with cerebral palsy.

The aim of this study was to examine the development of arithmetic performance and its cognitive precursors in children with CP from 7 till 9 years of age. Previous research has shown that children with CP are generally delayed in arithmetic performance compared to their typically developing peers. In children with CP, the developmental trajectory of the ability to solve addition- and subtraction tasks has, however, rarely been studied, as well as the cognitive factors affecting this trajectory. Sixty children (M=7.2 years, SD=.23 months at study entry) with CP participated in this study. Standardized tests were administered to assess arithmetic performance, word decoding skills, non-verbal intelligence, and working memory. The results showed that the ability to solve addition- and subtraction tasks increased over a two year period. Word decoding skills were positively related to the initial status of arithmetic performance. In addition, non-verbal intelligence and working memory were associated with the initial status and growth rate of arithmetic performance from 7 till 9 years of age. The current study highlights the importance of non-verbal intelligence and working memory to the development of arithmetic performance of children with CP.

Feasibility and reliability of the modified Berg Balance Scale in persons with severe intellectual and visual disabilities.

BACKGROUND: The purpose of this study was to determine the feasibility and reliability of the modified Berg Balance Scale (mBBS) in persons with severe intellectual and visual disabilities (severe multiple disabilities, SMD) assigned Gross Motor Function Classification System (GMFCS) grades I and II. METHOD: Thirty-nine participants with SMD and GMFCS grades I and II performed the mBBS twice with 1-week interval. Feasibility was assessed by the percentage of successful measurements per task and of the total score. First, test-retest reliability was determined by intraclass correlation coefficients (ICC) for each task and for the total score of all tasks combined. Second, level of agreement between test-retest scores was assessed with the proportion of equal scores for each task. Finally, internal consistency of the distinct tasks was assessed by Cronbach's alpha. RESULTS: The results indicated that 92% of the measurements by the mBBS for all selected tasks were successful, indicating that the mBBS is a feasible instrument for the tested target group. ICC for the test-retest of the total score was 0.95. The proportion of equal scores for test-retest of the tasks was 0.80 or more, except for tasks 9 and 10. Cronbach's alpha of distinct tasks was 0.84. Test-retest reliability of tasks 9 and 10 was not acceptable. CONCLUSIONS: Feasibility of all tasks and test-retest reliability of 10 out of 12 mBBS tasks is acceptable. The mBBS is a both feasible and reliable test for evaluating the functional balance of persons with SMD and GMFCS grades I and II.

Neural evidence for impaired action selection in right hemiparetic cerebral palsy.

Recent studies suggest that in addition to low-level motor impairments, individuals with hemiparetic cerebral palsy (HCP) are characterized by anticipatory action planning deficits as well. In the present EEG study we investigated the neural and temporal dynamics of action planning in participants with right-sided HCP (n=10) and in left-handed control subjects (n=10). An anticipatory planning task was used in which participants were required to grasp and rotate a hexagonal knob over different angles (60 degrees, 120 degrees or 180 degrees). At a behavioral level, participants with HCP were slower in their movements and often selected an inappropriate grip when grasping the object. At a neural level, individuals with HCP showed a strong reduction in the amplitude of the P2 component, likely reflecting an impaired process of action selection. In addition, a strong correlation was observed between the P2 amplitude and grasping and rotation times. The P2 component was localized to sources in the dorsal posterior cingulate cortex (dPCC), an area that is known to be involved in orienting visual body parts in space. Together these findings suggest that anticipatory planning deficits in cerebral palsy arise mainly due to an impaired process of action selection.

Discrete bimanual co-ordination in children and young adolescents with hemiparetic cerebral palsy: recent findings, implications and future research directions.

Bimanual co-ordination is a field that has generated much research interest. It is clear that when hands move simultaneously there is a tendency for such movements to be synchronized. This phenomenon has also been observed in individuals with congenital brain damage, such as cerebral palsy. This paper provides an overview of past and current work in the area of bimanual co-ordination looking at the nature of the coupling and the underlying control of such movements. Work on 'non-disabled' individuals and unimanual reaching and grasping is included, but the main emphasis is bimanual co-ordination in children with hemiparetic cerebral palsy. The direction for future research is discussed and consideration given to the therapeutic implications of such work.

The influence of object size on discrete bimanual co-ordination in children with hemiplegic cerebral palsy.

PURPOSE: To examine the influence of perturbation of object size on the nature and extent of interlimb coupling in children with hemiplegic cerebral palsy. METHOD: Participants completed a number of trials reaching and grasping unimanually and bimanually to a small cube (1.5 cm) and a large cube (6 cm). Both 3D kinematic data and video data were gathered and qualitative descriptions of the video data were made. RESULTS: It was found that object size did influence the nature and extent of bimanual coupling. As in previous studies this varied from participant to participant and either or a combination of temporal, spatial, or postural coupling was observed. In some cases the hemiplegic hand was influenced by the non hemiplegic hand, while in others it was the reverse. CONCLUSIONS: The influence of context and the individual nature of children with cerebral palsy observed in this paper must be considered by those in rehabilitation. Additionally, the therapist might be able to use the natural tendency to couple to assist the functional control of the hemiplegic side.

Shoulder and hand displacements during hitting, reaching, and grasping movements in hemiparetic cerebral palsy.

In this study, we examined the degree and timing of shoulder displacements during hitting, reaching, and grasping movements performed by young adults with hemiparetic cerebral palsy. The participants performed unimanual and bimanual arm movements towards targets and objects of different sizes. On the basis of the assumption that shoulder displacement due to trunk translation and rotation is a successful adaptive reaction to reduced joint mobility in the affected arm, the fluency of hand displacements was expected to remain invariant under variations of shoulder displacement as is also the case in healthy participants. The results point in this direction. With respect to the timing of shoulder displacement, prior research suggested that hemiparetic movements can be characterized by inconsistent motion-timing patterns-that is, the timing of the of shoulder and hand-displacement onsets varied between trials. Therefore, the within-subject variability of the movement-onset asynchrony between hand and ipsilateral shoulder displacement was expected to be larger on the impaired side than on the unimpaired side. This prediction was not confirmed, which challenges these earlier conclusions. Additionally, we also examined the peak-velocity asynchrony of the hand and shoulder. Contrary to the onset asynchrony, the peak asynchrony varied between the hitting and reaching task and between the hitting and grasping task. For the reaching and grasping tasks, there were also significant differences between hands. Again, variability of the (peak-velocity) asynchrony was not significantly increased when comparing the impaired hand with the unimpaired hand. The results suggests that the hemiparetic participants were capable of flexibly recruiting and sequencing the various degrees of freedom of their impaired side required for successful task completion, albeit in different magnitudes and sequenced differently.