Barriers to recruitment of children with cerebral palsy in a trial of home-based training.

Many trials fail to include the targeted number of participants, causing scientific and ethical problems. The COAD trial of home-based training programs (HBTPs) for children with unilateral cerebral palsy (CP) encountered recruitment problems, even though the parent-delivered home-based approach complies with recent health-care developments in the Netherlands. The current project aimed to identify the barriers to recruitment in the COAD trial. This summative, multidimensional evaluation comprised informal conversational interviews in which stakeholders who had been involved reflected on the factors that impeded successful recruitment of participants into the COAD trial. Barriers to implementation and recruitment were clustered according to the constructs of the Consolidated Framework for Implementation Research (CFIR). Member checking validated the findings. A total of 41 stakeholders contributed to the evaluation. Barriers to the implementation of the HBTPs were identified within every domain of the CFIR (intervention characteristics, outer setting, inner setting, characteristics of individuals, and process). Parent-delivered home-based training was perceived as highly complex and in conflict with the pressures on and the needs of parents. Many parents preferred the alternative center-based group interventions. The involvement of a resonance group was highly valued, and opportunities for further enhancements emerged. Additionally, the importance of research consortia was emphasized. The appropriateness of the RCT as the study design was criticized. The findings of this study are summarized in a tool which provides a dozen directions for the successful recruitment of participants in pediatric rehabilitation research.

The relationship between motor abilities and quality of life in children with severe multiple disabilities.

BACKGROUND: This study aimed to determine the relationship between motor abilities and quality of life in children with severe multiple disabilities. METHODS: In this cross-sectional study, motor abilities of 29 children (mean age 9.8 years; 45% girls) with severe multiple disabilities [IQ < 25; Gross Motor Function Motor Classification System level V] were measured with the MOtor eVAluation in Kids with Intellectual and Complex disabilities (Movakic) questionnaire (completed by the child's physical therapist). Quality of life was measured with the Quality of Life-Profound Multiple Disabilities (QoL-PMD) questionnaire (completed by the child's parents). RESULTS: A significantly moderate to high correlation was found between the total scores on the Movakic and the QoL-PMD (r = 0.40, P = 0.03), indicating that higher scores in motor abilities are associated with a higher level of quality of life. Furthermore, significantly moderate to high correlations were found between the total score on the Movakic and the dimension Physical Well-Being, Development and Activities of the Qol-PMD. In multiple linear regression models, all significant bivariate relationships between the Movakic total scores and QoL-PMD dimensions remained significant after controlling for the Gross Motor Function Motor Classification System level. CONCLUSIONS: In these children with severe multiple disabilities, motor abilities (as measured by Movakic) are moderately related to quality of life (as measured by the QoL-PMD).

Does intensive upper limb treatment modality Hybrid Constrained Induced Movement Therapy (H-CIMT) improve grip and pinch strength or fatigability of the affected hand?

PURPOSE: To investigate the effects of Hybrid-Constrained Induced Movement Therapy (H-CIMT), defined as CIMT combined with Bimanual Intensive Movement Therapy (BIMT), on grip and pinch strength and fatigability we measured grip and pinch strength and fatigability during clinical H-CIMT. METHODS: The children participated in a H-CIMT model organized in a therapeutic summer-camp. Children received 90 hours of intensive treatment. Grip and pinch strength and fatigability was measured and fatigue was calculated according to a Static Fatigue Index (SFI). RESULTS: Pinch strength significantly increased, grip strength did not increase significantly. A non-significant decrease was seen in SFI in pinch and grip. CONCLUSIONS: H-CIMT showed to be effective in increasing muscle pinch strength in the AH. Effectiveness in decreasing muscle fatigue during grip and pinch tests is not yet shown although there was a tendency towards a decrease in muscle fatigue. However, the long-term effects on these aspects are also important in future research.

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.

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.

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.

Efficacy of upper limb strengthening in children with Cerebral Palsy: A critical review.

OBJECTIVE: This review focuses on the effects of strengthening interventions of the upper limb in children with Cerebral Palsy (CP). The strengthening intervention studies were divided in two categories: those based on stand-alone strength training, and those on strength training combined with other interventions. DATA SOURCES AND EXTRACTION: A search in all relevant databases was performed. DATA SYNTHESIS: Six articles were included: three randomized controlled trials (RCTs), two clinical trial (CT) and one case study. Effect sizes of strength training on muscle strength and function of the upper limb were calculated. CONCLUSION: There are no coherent recommendations for strength training, based on these studies. The causes include too much variety of types of training, level of intensity and duration. All of the reported upper limb strength training studies found an increase in muscle strength. In addition, the quality of these studies was not high. More RCTs on strength training according to the official strength training guidelines are necessary to assess the impact and potential of strength training of the upper limb to improve the daily activities and participation in children with CP.

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.

Children with generalised joint hypermobility and musculoskeletal complaints: state of the art on diagnostics, clinical characteristics, and treatment.

INTRODUCTION: To provide a state of the art on diagnostics, clinical characteristics, and treatment of paediatric generalised joint hypermobility (GJH) and joint hypermobility syndrome (JHS). METHOD: A narrative review was performed regarding diagnostics and clinical characteristics. Effectiveness of treatment was evaluated by systematic review. Searches of Medline and Central were performed and included nonsymptomatic and symptomatic forms of GJH (JHS, collagen diseases). RESULTS: In the last decade, scientific research has accumulated on all domains of the ICF. GJH/JHS can be considered as a clinical entity, which can have serious effects during all stages of life. However research regarding the pathological mechanism has resulted in new potential opportunities for treatment. When regarding the effectiveness of current treatments, the search identified 1318 studies, from which three were included (JHS: n = 2, Osteogenesis Imperfecta: n = 1). According to the best evidence synthesis, there was strong evidence that enhancing physical fitness is an effective treatment for children with JHS. However this was based on only two studies. CONCLUSION: Based on the sparsely available knowledge on intervention studies, future longitudinal studies should focus on the effect of physical activity, fitness, and joint stabilisation. In JHS and chronic pain, the effectiveness of a multidisciplinary approach should be investigated.

Botulinum toxin-a in children with congenital spastic hemiplegia does not improve upper extremity motor-related function over rehabilitation alone: a randomized controlled trial.

BACKGROUND: Rehabilitation of the upper extremity in children with hemiplegic cerebral palsy has not been compared to the same intensity of therapy combined with injected botulinum toxin (BTX). OBJECTIVE: To measure the short-term (2 weeks) and long-term (6 and 9 months) effects of a standardized functional training program versus without the addition of chemodenervation of forearm and hand muscles. METHODS: Twenty children with spastic hemiplegia, aged 4 to 16 years, were matched for baseline characteristics and then randomized to standardized functional physical and occupational therapies for 6 months (PT/OT group) or to the same therapies plus multimuscle BTX-A (BTX+ group). MAIN OUTCOME MEASURES: were isometric generated force, overshoot and undershoot (force production error), active and passive range of motion by goniometry (ROM), stretch restricted angle (SRA) of joints, Ashworth scores at the elbow and wrist, and the Melbourne assessment of unilateral upper limb function. All measures were performed at baseline, 2 weeks after BTX-A, 6 months (end of therapy), and then 3 months after termination of the therapy. RESULTS: Clinical measures (muscle tone, active ROM of wrist and elbow) showed improvement in both groups. However, no significant differences emerged between groups on functional measures. Generated force decreased directly after the BTX-A injection but increased during the therapy period. The PT/OT group, however, showed a significantly higher increase in force and accuracy with therapy compared with the BTX+ therapy group. CONCLUSIONS: Functional rehabilitation therapies for the upper extremity increase manual isometric flexor force at the wrist and ROM, but BTX injections cause weakness and do not lead to better outcomes than therapy alone.

Children with congenital spastic hemiplegia obey Fitts' Law in a visually guided tapping task.

Fitts' Law is commonly found to apply to motor tasks involving precise aiming movements. Children with cerebral palsy (CP) have severe difficulties in such tasks and it is unknown whether they obey Fitts' Law despite their motor difficulties. If Fitts' Law still does apply to these children, this would indicate that this law is extremely robust and that even performance of children with damaged central nervous systems can adhere to it. The integrity of motor control processes in spastic CP is usually tested in complex motor tasks, making it difficult to determine whether poor performance is due to a motor output deficit or to problems related to cognitive processes since both affect movement precision. In the present study a simple task was designed to evaluate Fitts' Law. Tapping movements were evaluated in 22 children with congenital spastic hemiplegia (CSH) and 22 typically developing children. Targets (2.5 and 5 cm in width) were placed at distances of 10 and 20 cm from each other in order to provide Indices of Difficulty (ID) of 2-4 bits. Using this Fitts' aiming task, prolonged reaction and movement time (MT) were found in the affected hand under all conditions in children with CSH as compared to controls. Like in the control group, MT in children with CSH was related to ID. The intercept 'a', corresponding to the time required to realize a tapping movement, was higher in the affected hand of the children in the CSH group. Although, the slope b (which reflects the sensitivity of the motor system to a change in difficulty of the task) and the reciprocal of slope (that represents the cognitive information processing capacity, expressed in bits/s) were similar in both groups. In conclusion, children with CSH obey Fitts' Law despite very obvious limitations in fine motor control.

Late developmental deficits in force control in children with hemiplegia.

Differences related to development were investigated using a finger isometric force task in children with cerebral palsy and control children. The increase in force and force control observed in controls did not take place in patients. In the younger subset of patients (< 10 years) the force produced by the non-affected hand was greater than in either hand of young controls. This effect was not observed in the older subset of patients (< 10 years). Older controls also differed from younger controls in that they used higher frequency feedback loops. In contrast, older patients failed to show this increase. Moreover, the failure occurred both in the affected and non-affected hand, indicating that abnormalities involve the force control system of both sides.