Petra E. M. van Schie

Diffusion-weighted and Conventional MR Imaging in Neonatal Hypoxic Ischemia: Two-year Follow-up Study

PURPOSE To establish the supplemental value of diffusion-weighted (DW) magnetic resonance (MR) imaging beyond conventional MR to predict clinical outcome after neonatal hypoxic ischemia (HI) at 2 years of age. MATERIALS AND METHODS Forty-six infants with neonatal HI were enrolled in this prospective study, after approval by the local ethical committee and informed consent of the parents. Neonatal MR imaging ranged from 1 to 45 days after birth. Apparent diffusion coefficient (ADC) was measured in 14 brain regions. DW and conventional images were qualitatively scored for abnormalities, resulting in cumulative scores and patterns of damage. Surviving infants were scored for motor outcome at the age of 2 years, and outcome was classified as poor if the motor score was less than 70 or in case of death. Analyses were performed for the whole group, with additional analyses for the early (0-4 days after birth) and late (>4 days after birth) imaging groups. RESULTS Twenty-five infants had a good outcome and 21 had a poor outcome. Only in the early imaging group, the infants with poor outcome had significantly lowered ADC values in several brain areas, with the posterior limb of the internal capsule being the most predictive (Wald score = 5.7; P = .017). Cumulative scores of DW imaging were the best predictor of poor motor outcome at the age of 2 years (Wald score = 7.2, P < .01). The basal ganglia and central cortex and the diffuse pattern of brain damage were highly associated with poor outcome (Fisher exact test = 29.8; P < .001). CONCLUSION In neonatal HI, DW imaging is a useful additional MR technique to predict the motor outcome at 2 years. Local ADC values had a limited value. Recognition of the patterns of brain damage with DW and conventional MR imaging can be used as a diagnostic tool in neonatal HI.

Short- and long-term effects of selective dorsal rhizotomy on gross motor function in ambulatory children with spastic diplegia

OBJECT The primary aim of this prospective cohort study was to evaluate the short-term (1 year) and long-term (mean 6 years) effects of selective dorsal rhizotomy (SDR) on gross motor function and spasticity in ambulatory children with spastic diplegia. Secondary aims were to investigate side effects, additional treatment during follow-up (botulinum toxin type A injections or orthopedic surgery), and parental satisfaction. METHODS Thirty-three children who had undergone SDR at a mean age of 6 years and 7 months (± 2 years) were included. There were 7 children at Gross Motor Function Classification System (GMFCS) Level I, 7 at Level II, and 19 at Level III. Gross motor function was assessed with the Gross Motor Function Measure-66 (GMFM-66). Spasticity was measured according to a modified Tardieu scale. Side effects, additional treatment, and parental satisfaction were recorded using a parental questionnaire and medical records. RESULTS At 1-year follow-up, mean GMFM-66 scores improved significantly by 4.3 ± 4.1 points. Children at GMFCS Levels I and II showed significantly more improvement (7.2 points) on the GMFM-66 compared with children at GMFCS Level III (2.9 points). On long-term follow-up (mean 6 years ± 22 months), mean GMFM-66 scores improved significantly by 6.5 ± 5.9 points, without a difference between children at GMFCS Levels I and II and Level III. No relapse of spasticity was noted. Ten children (30%) needed orthopedic surgery and 13 children (39%) received botulinum toxin type A treatment after SDR. Twenty (91%) of the 22 parents who answered the questionnaire at long-term follow-up believed that their childs functioning had improved after SDR. CONCLUSIONS Selective dorsal rhizotomy resulted in short- and long-term improvements in gross motor function, without relapse of spasticity. However, the majority of the children still needed additional surgery or botulinum toxin A treatment.

Family-centred services in the Netherlands: validating a self-report measure for paediatric service providers

Objective: To validate the Dutch translation of the Canadian Measure of Processes of Care for Service Providers questionnaire (MPOC-SP) for use in paediatric rehabilitation settings in the Netherlands. Design: The construct validity, content validity, face validity, and reliability of the Dutch MPOC-SP were determined. Subjects: The 163 service providers that participated in the validation study represented seven childrens rehabilitation centres and affiliated schools in the Netherlands (overall response rate 55.6%). In this sample 19 disciplines were represented. Main measures: The MPOC-SP consists of 27 items (assessing four domains) and was designed to examine how service providers think about the quality of care they provide and to assess the extent to which these services are family centred. Fifty-three service providers filled out an additional face validity questionnaire. Results: All items correlated best and significantly with their own scale score (rs 0.48-0.82, P < 0.001). The Pearsons correlation coefficients were all significant and confirmed that the four scales measure different aspects of a same construct, namely family-centred service. The content validity and the face validity of the Dutch MPOC-SP were good, indicating the questionnaire measures relevant aspects of family-centred service delivery in paediatric rehabilitation settings in the Netherlands. The test-retest analyses (intraclass correlation coefficient (ICC) 0.83-0.89) and the internal consistency analyses (alpha 0.65-0.84) showed that the Dutch MPOC-SP is a reliable tool. Conclusions: The Dutch MPOC-SP is a reliable and valid instrument to measure the family-centredness of service delivery.

Quality of paediatric rehabilitation from the parent perspective: validation of the short Measure of Processes of Care (MPOC-20) in the Netherlands

Objective: In the present study we aim to assess the reliability and validity of the 20-item version of the Dutch Measure of Processes of Care (MPOC). Design: The reliability, concurrent validity, predictive validity and construct validity of the Dutch MPOC-20 were determined. A subset of MPOC-20 data was extracted from a large Dutch MPOC (56-item version) database. Subjects: Participants were 405 mothers and 22 fathers of children aged 1-18 years recruited through nine paediatric rehabilitation centres in the Netherlands. Main measures: The participants filled out the MPOC-20 items, the Client Satisfaction Questionnaire (CSQ), and two additional questions about satisfaction with services and the amount of stress they experienced. Results: The internal consistency analyses (alphas 0.75-0.87) and the test-retest analyses (intraclass correlation coefficients (ICCs) 0.78-0.91) showed that the Dutch MPOC-20 is a reliable tool. The concurrent validity of the Dutch MPOC-20 was confirmed by positive correlations between MPOC-20 scale scores and the CSQ (r 0.39-0.69), and between MPOC-20 scale scores and an overall satisfaction variable (r 0.37-0.66). The predictive validity of the Dutch MPOC-20 was supported by moderately negative correlations between MPOC-20 scores and a stress variable (r -0.27 to -0.44). The construct validity of the Dutch MPOC-20 was confirmed by significant scale intercorrelations (r 0.41-0.84) and a factor analysis. Conclusions: The 20-item version of the MPOC (Dutch MPOC-20) is a reliable and valid measure of the family-centredness of paediatric rehabilitation.

Developmental trajectories of receptive and expressive communication in children and young adults with cerebral palsy.

The aim of this study was to determine the developmental trajectories of expressive (speech) and receptive (spoken and written language) communication by type of motor disorder and intellectual disability in individuals with cerebral palsy (CP).

Long-term effect of selective dorsal rhizotomy on gross motor function in ambulant children with spastic bilateral cerebral palsy, compared with reference centiles

The aim of this study was to evaluate the long‐term effect of selective dorsal rhizotomy (SDR) on the gross motor function of ambulant children with spastic bilateral cerebral palsy (CP), compared with reference centiles.

Effects of intrathecal baclofen on daily care in children with secondary generalized dystonia: A pilot study

AIM Treatment options for dystonic cerebral palsy (CP) are limited. Our aims were to determine whether intrathecal baclofen (ITB) improves daily care, decreases dystonia and decreases pain in patients with dystonic CP. METHODS Patients received randomized blinded treatment with ITB or placebo. Scores on problems of daily care were recorded and dystonia, pain and comfort were assessed. RESULTS Four patients (three males, average age 12 years 6 months) were included (all Gross Motor Function Classification System level V). During the trial period problem scores and dystonia scores decreased in all four patients. CONCLUSION In this pilot study we report positive functional effects of ITB trial treatment in four patients with dystonic CP. A randomized trial with a larger cohort is needed to verify these results.

How Do Changes in Motor Capacity, Motor Capability, and Motor Performance Relate in Children and Adolescents With Cerebral Palsy?

OBJECTIVE To investigate the relations between changes in motor capacity (can do, in standardized environment), motor capability (can do, in daily environment), and motor performance (does do, in daily environment) among children with cerebral palsy (CP). DESIGN Prospective longitudinal study. After baseline measurements (at the age of 18 mo, 30 mo, 5 y, 7 y, 9 y, 11 y, or 13 y), 2-year follow-up measurements were performed. Change scores were calculated, and Pearson correlations were used for change score relations. SETTING Outpatient clinic. PARTICIPANTS Toddlers, school-age children, and adolescents with CP (N=321; 200 boys, 121 girls). Levels of severity according to the Gross Motor Function Classification System included level I (42%), level II (15%), level III (17%), level IV (13%), and level V (13%). INTERVENTIONS Not applicable. MAIN OUTCOME MEASURES Change in motor capacity was assessed with the Gross Motor Function Measure-66. Changes in motor capability and motor performance were assessed with the Pediatric Evaluation of Disability Inventory using the Functional Skills Scale and Caregiver Assistance Scale, respectively. RESULTS Within the total group, change score correlations were moderate (.52-.67) and significant (P<.001). For age groups, correlations were significantly higher in toddlers than school-age children and adolescents. For severity levels, correlations were significantly higher in children at level III than level I, IV, and V. CONCLUSIONS Results imply that change in motor capacity does not automatically translate to change in motor capability and change in motor capability does not automatically translate to change in motor performance. Results also show different relations for clinically relevant subgroups. These are important insights for clinical practice because they can guide evidence-based interventions with a focus on activities.

Validation of the Dutch Giving Youth a Voice Questionnaire (GYV-20): a measure of the client-centredness of rehabilitation services from an adolescent perspective.

Purpose. The objective was to validate the Dutch translation of the Canadian Giving Youth a Voice Questionnaire (GYV-20) for use in paediatric rehabilitation settings in The Netherlands. The GYV-20 consists of 20 items (assessing four domains) and was designed to evaluate the client-centredness of rehabilitation services from an adolescent perspective. Method. The construct validity, concurrent validity, and reliability of the Dutch GYV-20 were determined. Participants were 116 youngsters aged 11 – 21 years (Mean = 15.9; SD = 2.1) recruited through six paediatric rehabilitation settings in The Netherlands. Results. Correlations between the GYV-20 scale scores were positive (r = 0.69 – 0.78). The GYV-20 showed adequate internal consistency, with Cronbachs α ranging from 0.76 – 0.81. The ICCs of test-retest reliability ranged from 0.82 – 0.92, which demonstrated good stability of the GYV-20. Dutch adolescents judged the GYV-20 as a valuable and useful tool to evaluate rehabilitation services in The Netherlands. Conclusions. The Dutch GYV-20 showed sufficient evidence of construct validity and good reliabilities. The Dutch GYV-20 offers users a useful measurement option for various research and clinical purposes.

Intrathecal baclofen for progressive neurological disease in childhood: A systematic review of literature

BACKGROUND Intrathecal baclofen (ITB) treatment is frequently used for individuals with severe, but non-progressive, spasticity refractory to oral treatment. However, experiences with ITB in patients with progressive neurological disorders of childhood causing spasticity are limited. AIM To investigate whether ITB is an option in patients with progressive neurological disorders causing spasticity in childhood. DESIGN A systematic literature search in Embase, Pubmed and the Cochrane Library was performed. RESULTS We identified six eligible studies considering patients with progressive neurological disease in childhood and receiving ITB treatment. The studies included a total of seven paediatric patients and four adult patients. Improvement was reported in spasticity, spasms, pain, gait, activities of daily life and providing care. High satisfaction is described. CONCLUSIONS ITB has beneficial effects in paediatric patients with progressive neurological disease. However, the level of evidence is limited due to the small number of available studies and due to the poor quality of these studies.