Jan Willem Gorter

Early Autism Detection: Are We Ready for Routine Screening?

BACKGROUND. Autism is a serious neurodevelopmental disorder that has a reportedly rising prevalence rate. The American Academy of Pediatrics recommends that screening for autism be incorporated into routine practice. It is important to consider the pros and cons of conducting autism screening as part of routine practice and its implications on the community. We have explored this question in the context of screening from a scientific point of view. METHOD: A literature search was conducted to assess the effectiveness of community screening programs for autism. RESULTS: Judged against critical questions about autism, screening programs failed to fulfill most criteria. Good screening tools and efficacious treatment are lacking, and there is no evidence yet that such a program would do more good than harm. CONCLUSIONS: On the basis of the available research, we believe that we do not have enough sound evidence to support the implementation of a routine population-based screening program for autism. Ongoing research in this field is certainly needed, including the development of excellent screening instruments and demonstrating with clinical trials that such programs work and do more good than harm.

Goal attainment scaling in paediatric rehabilitation: a critical review of the literature

The aim of the study was to review the psychometric properties and use of goal attainment scaling (GAS) in paediatric rehabilitation research. We performed a critical literature review searching: (1) all studies whose main focus was to assess the psychometric properties of GAS in paediatric rehabilitation; and (2) all effect studies in paediatric rehabilitation that used GAS as one of the outcome measures. Three articles in the first group and six in the second group met the inclusion criteria. None of the studies had investigated the content reliability of the scales. Interrater reliability had been investigated in one study and had been found to be good. Only one trial had assessed the content validity of the developed scales, which was found to be acceptable. Comparisons showed that GAS, because of its idiosyncratic nature, measures different constructs from those measured by some related instruments. Low concurrent validity was found. All included studies reported good sensitivity to change. We conclude that the literature supports promising qualities of GAS in paediatric rehabilitation. GAS is a responsive method for individual goal setting and for treatment evaluation. However, current knowledge about its reliability when used with children is insufficient. There is a need for further development of GAS and its application for children of different ages and disabilities, across therapists of different disciplines.

Capacity, Capability, and Performance: Different Constructs or Three of a Kind?

OBJECTIVES The present study focused on motor activities of young children with cerebral palsy (CP) and examined the relation between motor capacity (what a person can do in a standardized, controlled environment), motor capability (what a person can do in his/her daily environment), and motor performance (what a person actually does do in his/her daily environment). DESIGN The relations between motor capacity, motor capability, and motor performance were calculated by using Pearson correlations and visualized by scatterplots. SETTING A cross-sectional study of a hospital-based population of children with CP. PARTICIPANTS Subjects were children with CP (N=85) aged 30 months (Gross Motor Function Classification System levels I-V). INTERVENTIONS Not applicable. MAIN OUTCOME MEASURES Motor capacity, motor capability, and motor performance were assessed with the Gross Motor Function Measure and 2 scales of the Pediatric Evaluation of Disability Inventory, respectively. RESULTS Correlations between motor capacity, motor capability, and motor performance were high, between 0.84 and 0.92, and significant (P< .001). But when comparing children with the same level of motor capacity or motor capability, large ranges at the level of motor performance were found. CONCLUSIONS Results imply that motor performance levels are only partly reflected by the motor capacity and motor capability levels in young children children with CP. Contextual factors (physical and social environment) and personal factors (such as motivation) influence the relations between capacity, capability, and performance. This information is essential in making decisions about the focus of therapy to maximize a childs independent functioning in daily life.

Decreasing Incidence and Severity of Cerebral Palsy in Prematurely Born Children

OBJECTIVE To examine incidence and severity of cerebral palsy (CP), and associated factors among preterm survivors (gestational age <34 weeks), admitted to a neonatal intensive care unit from 1990-2005. STUDY DESIGN Eighteen antenatal, perinatal and postnatal factors were analyzed. The cohort was divided in four birth periods: 1990-1993 (n=661), 1994-1997 (n=726), 1998-2001 (n=723), and 2002-2005 (n=850). The Gross Motor Function Classification System was used as primary outcome measure (mean age: 32.9 ± 5.3 months). Logistic regression analyses were used. RESULTS CP incidence decreased from 6.5% in period I, to 2.6%, 2.9% and 2.2% (P<.001) in period II-IV, respectively. Simultaneously, cystic periventricular leukomalacia (c-PVL) decreased from 3.3% in period I to 1.3% in period IV (P=.004). Within the total cohort (n=3287), c-PVL grade III decreased from 2.3% in period I to 0.2% in period IV (P=.003). The number of children with Gross Motor Function Classification System levels III-V decreased from period I to IV (P=.035). Independent risk factors for CP were c-PVL and severe intraventricular hemorrhage, whereas antenatal antibiotics, presence of an arterial line, Caesarean section, and gestational age were independent protective factors. CONCLUSION CP incidence and severity decreased from 1990-1993 onward, which could be attributed to a reduction of 93% in severe c-PVL.

Use of the GMFCS in infants with CP: the need for reclassification at age 2 years or older

The stability of the Gross Motor Function Classification System (GMFCS) over time is described in 77 infants (41 boys, 36 girls) with cerebral palsy (CP; mean age 19.4mo [SD 1.6 mo]; 27 unilateral spastic, 42 bilateral spastic, eight dyskinetic type) and in the same children at follow‐up at age 2 to 4 years. The overall level of agreement over time (linear weighted kappa) was 0.70 (95% confidence interval [CI] 0.61−0.79). The overall percentage of children whose GMFCS level changed one or two levels was 42%, of which the majority were reclassified to a less functional level (McNemar’s Chi2 test p=0.11). The chance that children initially classified in the combination of GMFCS Levels I, II, and III would subsequently be classified in the same level in early childhood was 96% (positive predictive value [PPV] 0.96, 95% CI 0.85−0.99), whereas the PPV for the combination of Levels I and II was 0.88, 95% CI 0.70−0.96. These findings indicate that GMFCS classification in infants is less precise than classification over time in older children. In conclusion, children can be classified by the GMFCS early on, but there is a need for reclassification at age 2 or older as more clinical information becomes available.

Reliability for Running Tests for Measuring Agility and Anaerobic Muscle Power in Children and Adolescents with Cerebal Palsy

Purpose: We investigated reliability, construct validity, and feasibility of two sprint tests for children with cerebral palsy (CP). Methods: Twenty-six children with CP participated (7–18 years of age; Gross Motor Function Classification System [GMFCS] level I or II). On different occasions, the 10 × 5-Meter Sprint Test and the Muscle Power Sprint Test were scored by different assessors. Results: Excellent interobserver reliability (intraclass correlation [ICC] = 1.0 and ICC ≥ 0.97) and test−retest reliability (ICC = 0.97 and ICC ≥ 0.97) were obtained. Scores differed significantly on both sprint tests for children classified at GMFCS level I and level II. Mean scores for feasibility ranged from 8.8 to 9.2 on a 10-cm visual analog scale (10 = the best). Conclusions: Both exercise tests are reliable and have good feasibility for children and adolescents with CP (GMFCS level I or II). Construct validity is supported for both sprint tests in children classified at GMFCS level I and level II.

Cross-cultural validation and psychometric evaluation of the Dutch language version of the Children’s Assessment of Participation and Enjoyment (CAPE) in children with and without physical disabilities

Objective: To assess construct validity, test—retest reliability and inter-interviewer reliability of the intensity dimension of the Children’s Assessment of Participation and Enjoyment (CAPE) for children with and without physical disabilities in the Netherlands. Subjects: In total, 232 children aged 6—18 years (110 male, 122 female) participated. Seventy-four children with various physical disabilities and 158 without a disability. Design: Participants completed the CAPE and the Peabody Picture Vocabulary Test. In addition, parents of 142 children were interviewed using the Vineland Adaptive Behavior Scales and the Family Environment Scale. For 71 children test— retest reliability and for 60 children inter-interviewer reliability were assessed. Validity was examined by assessing differences in participation intensity in children with disabilities versus without a physical disability, boys versus girls, and younger versus older children. In addition, 13 hypotheses regarding participation, child and family variables were examined. Results: Validity of the CAPE was supported by significant differences in participation for subgroups. Participation differed significantly in children with and without disabilities. Girls participated more in all activities. Older children participated more in social activities and self-improvement activities, younger children participated more in recreational activities. Validity of the CAPE was further supported with significant correlation coefficients in 8 out of 13 hypotheses. Both test—retest and inter-interviewer reliability were good to excellent. Conclusions: The findings indicate that the Dutch language version of the CAPE is a reliable and valid instrument to measure participation in recreation and leisure activities for children with and without physical disabilities aged 6—18 years.

Responsiveness of Goal Attainment Scaling in comparison to two standardized measures in outcome evaluation of children with cerebral palsy

Objectives: To assess the responsiveness of Goal Attainment Scaling compared with the Pediatric Evaluation of Disability Inventory (PEDI) and the 66-item Gross Motor Function Measure (GMFM-66) in multidisciplinary rehabilitation practice. Design: Observational study. Pretest–posttest design. Subjects/patients: Twenty-three children with cerebral palsy, aged 2–13 years. Methods: Goal Attainment Scaling, PEDI and GMFM-66 assessments were performed before and after six months of treatment. Physical, occupational and speech therapists constructed and scored 6-point Goal Attainment Scaling scales meeting predetermined criteria, describing the main functional goal per discipline. The contents of the three measures were compared using International Classification of Functioning, Disability and Health child and youth version (ICF-CY) codes. Spearman’s rho correlations between Goal Attainment Scaling change scores per discipline and change scores obtained with the PEDI functional skills scales and GMFM-66 were calculated. Complete goal attainment was compared with significant change in terms of the standardized measures. Results: Twenty per cent of the Goal Attainment Scaling items were not covered by items of the PEDI or the GMFM-66. Inconclusive correlations were found between Goal Attainment Scaling and PEDI change scores (r 0.28–0.64). Even after exclusion of the non-corresponding items, correlations were moderate (r 0.57–0.73). Of 39/64 Goal Attainment Scaling scales scored as complete goal attainment, 16 individual PEDI scores did not show change on the corresponding scale. Low correlation was found between Goal Attainment Scaling change scores and GMFM-66 change scores. Conclusion: Goal Attainment Scaling, PEDI and GMFM-66 were complementary in their ability to measure individual change over time in children with cerebral palsy. Using only the standardized instruments could have caused many individual rehabilitation goals actually attained being missed in the outcome evaluation.

Parents' reactions to the diagnosis of cerebral palsy: associations between resolution, age and severity of disability

BACKGROUND For parents, receiving a diagnosis, typically in early childhood, that their child has cerebral palsy may conjure up high distress and anxiety. Resolution of these initial reactions may help parents to focus on the challenges and needs of their children. AIMS of the study were to test whether parents of older children displayed resolution more often than parents of younger children, and whether parents of children with less severe cerebral palsy also showed more resolution. METHOD Resolution of reactions to diagnosis was assessed with the Reaction to Diagnosis Interview, in a clinic-based sample of 255 parents of children with cerebral palsy aged between 1.4 and 17.3 years. Physicians rated motor ability using the Gross Motor Function Classification System. RESULTS Overall, the responses of 81.6% of the parents were predominantly indicative of resolution. Unresolved reactions were significantly more often found among parents of younger children and parents of children with more severe motor disabilities. Among parents of teenage children, resolution was more often apparent from a focus on action to better the lives of their children, whereas in parents of younger children, it was more apparent from their focus on constructive thoughts and information seeking. CONCLUSIONS Given time, the large majority of parents may resolve their reactions to the diagnosis that their child has cerebral palsy. Parents of the most severely affected children may need specific support which, given the age trends, might be aimed at different resolution processes for parents of younger and older children.

Physical activity in young children with cerebral palsy

Purpose. The aim of this study was to describe the physical activity levels of 5- and 7-year-old children with cerebral palsy (CP, n = 97), to compare their physical activity levels with those of typically developing peers (TD, n = 57) and the Dutch recommendation for physical activity, and to investigate the associated factors. Method. The level of physical activity (hours spent on sports and physical activity per week) and contextual factors were assessed with standardised questionnaires. Results. Mean duration of self-reported physical activity for children with CP was 3.4 (± 1.9) h/week, which was significantly less than the 5.8 (± 2.3) h/week for TD-peers. Ninety-three percent of the children with CP were insufficiently physically active according to the Dutch recommendation for physical activity. Multiple regression analyses showed that younger age and lower educational level of the mother were significantly associated with lower levels of physical activity for children with CP, while severity of CP was not associated with physical activity levels. Twenty-two percent of the parents reported that more facilities in sport and games are required for children with CP. Conclusion. Physical activity is low in young children with CP and needs to be promoted at an early stage.