The Pediatric Autism Research Cohort (PARC) Study: protocol for a patient-oriented prospective study examining trajectories of functioning in children with autism.

INTRODUCTION: The developmentally variable nature of autism poses challenges in providing timely services tailored to a child's needs. Despite a recent focus on longitudinal research, priority-setting initiatives with stakeholders highlighted the importance of studying a child's day-to-day functioning and social determinants of health to inform clinical care. To address this, we are conducting a pragmatic multi-site, patient-oriented longitudinal investigation: the Pediatric Autism Research Cohort (PARC) Study. In young children (<7 years of age) newly diagnosed with autism, we will: (1) examine variability in trajectories of adaptive functioning from the point of diagnosis into transition to school; and (2) identify factors associated with trajectories of adaptive functioning. METHODS AND ANALYSIS: We aim to recruit 1300 children under 7 years of age with a recent (within 12 months) diagnosis of autism from seven sites: six in Canada; one in Israel. Participants will be followed prospectively from diagnosis to age 8 years, with assessments at 6-month intervals. Parents/caregivers will complete questionnaires administered via a customized online research portal. Following each assessment timepoint, families will receive a research summary report describing their child's progress on adaptive functioning and related domains. Analysis of the longitudinal data will map trajectories and examine child, family and service characteristics associated with chronogeneity (interindividual and intraindividual heterogeneity over time) and possible trajectory turning points around sensitive periods like the transition to school. ETHICS AND DISSEMINATION: Ethics approvals have been received by all sites. All parents/respondents will provide informed consent when enrolling in the study. Using an integrated knowledge translation approach, where stakeholders are directly engaged in the research process, the PARC Study will identify factors associated with trajectories of functioning in children with autism. Resulting evidence will be shared with government policy makers to inform provincial and national programs. Findings will be disseminated at conferences and published in peer-reviewed journals.

Investigating the Associations Between Child Autistic Symptoms, Socioeconomic Context, and Family Life: A Pilot Study.

Objective: The day-to-day experience of families with an Autistic child may be shaped by both, child characteristics and available resources, which often are influenced by the socioeconomic context of the family. Using a socioecological approach, this study explored the quantitative associations between child autistic symptoms, family socioeconomic status, and family life. Methods: Data came from the Pediatric Autism Research Cohort-PARC Study (pilot). Parents of children with a recent diagnosis of autism completed a set of assessments, including the Autism Family Experience Questionnaire, Autism Impact Measure, and a Sociodemographic Questionnaire. A series of multiple, iterative linear regression models were constructed to ascertain quantitative associations between child autistic symptoms, socioeconomic context, and family life. Results: A total of 50 children (mean age: 76 months; SD: 9.5 months; and 84% male) with data on the variables of interest were included in the analysis. The frequency of child autistic symptoms was associated with family life outcomes (p = 0.02 and R 2 = 24%). Once autistic symptom frequency, symptom impact, and sociodemographic variables were considered, parents of higher educational attainment reported worse family life outcomes compared to their lesser-educated counterparts. This cumulative regression model had considerable explanatory capability (p = 0.01, R 2 = 40%). Conclusion: This study demonstrates the utility of using a socioecological approach to examine the dynamic interplay between child characteristics and family circumstances. Our findings suggest that family life for parents (of an autistic child) who have obtained higher education is reported (by the parents themselves) as less satisfactory compared to that of parents without higher education, once adjusted for the autistic symptom frequency of child, symptom impact, and income. These findings can inform the design and delivery of more family-centered care pathways during the years following a diagnosis of autism.

Health-related quality of life for extremely low birth weight adolescents in Canada, Germany, and the Netherlands.

OBJECTIVE: The goal was to compare health-related quality of life of 12- to 16-year-old adolescents born at an extremely low birth weight in regional cohorts from Ontario (Canada), Bavaria (Germany), and the Netherlands. METHODS: Patients were extremely low birth weight survivors from Canada, Germany, and the Netherlands. Health-related quality of life was assessed with Health Utilities Index 3. Missing data were substituted by proxy reports. Differences in mean Health Utilities Index 3 scores were tested by using analysis of variance. Differences in the numbers of children with affected attributes were tested by using logistic regression analyses. RESULTS: Survival rates were similar; response rates varied between 71% and 90%. Significant differences in health-related quality of life were found between the cohorts, with Dutch children scoring highest on Health Utilities Index 3 and German children scoring lowest, independent of birth weight, gestational age, and cerebral palsy. Differences in mean utility scores were mainly attributable to differences in the cognition health attribute. Most of the results were corroborated by logistic regression analyses. CONCLUSIONS: There were significant differences between the 3 cohorts in health-related quality of life, not related to differences in birth weight, gestational age, or cerebral palsy. Survival and response rates alone cannot explain these differences.

Self-perceived health-related quality of life of former extremely low birth weight infants at young adulthood.

OBJECTIVES: The goals were to compare the self-reported, health-related quality of life of former extremely low birth weight and normal birth weight infants at young adulthood and to determine whether there were any changes over time. METHODS: A prospective, longitudinal, population-based study with concurrent control subjects was performed. We interviewed 143 of 166 extremely low birth weight survivors (birth weight: 501-1000 g; 1977-1982 births) and 130 of 145 sociodemographically comparable, normal birth weight, reference subjects. Neurosensory impairments were present for 27% extremely low birth weight and 2% normal birth weight young adults. Health Utilities Index 2 was used to assess health status, and standard gamble technique was used to measure directly the self-reported, health-related, quality of life and 4 hypothetical health states. RESULTS: Extremely low birth weight young adults reported more functional limitations in cognition, sensation, mobility, and self-care, compared with control subjects. There were no differences between groups in the mean self-reported, health-related, quality of life or between impaired (n = 38) and nonimpaired (n = 105) extremely low birth weight subjects. However, with a conservative approach of assigning a score of 0 for 10 severely disabled, extremely low birth weight subjects, the mean health-related quality of life was significantly lower than control values. Repeated-measures analysis of variance to compare health-related quality-of-life measurements obtained for young adults and teens showed the same decline in scores over time for both groups. There were no differences between groups in the ratings provided for the hypothetical health states. CONCLUSIONS: At young adulthood, health-related quality of life was not related to size at birth or to the presence of disability. There was a small decrease in health-related quality-of-life scores over time for both groups.

School-age outcomes in children who were extremely low birth weight from four international population-based cohorts.

OBJECTIVE: The aim of this study was to determine whether learning and school problems in extremely low birth weight (ELBW) and reference children differ between cohorts in different countries. METHODS: Participants were 4 international population-based cohorts of ELBW survivors who were 500 to 1000 g birth weight from New Jersey, central-west Ontario, Bavaria, and Holland (n = 532) and were followed longitudinally from birth. Psychometric data were collected independently and prospectively and included at least 1 measure of cognitive status and 1 measure of achievement administered to the children between the ages of 8 and 11 years. Adjustments were made for comparison of all measures based on reference norms within each country. Information on special educational assistance and grade repetition was obtained from the parents. RESULTS: The overall follow-up rate was 84% (range: 74%-90%; n = 436). The proportion of children who performed within the normal range (> or =85) were as follows: IQ between 44% and 62%; reading between 46% and 81%; arithmetic between 31% and 76%; and spelling between 39% and 65%. Children from New Jersey had the lowest rates of cognitive and achievement deficits, and Bavarian children did less well in achievement scores relative to their peers and other cohorts. Despite these differences, more than half of all cohorts required special educational assistance and/or repeated a grade. CONCLUSIONS: School difficulties were found to be a serious sequelae of ELBW in all 4 countries, an observation that has social and economic implications.

Psychopathology and social competencies of adolescents who were extremely low birth weight.

BACKGROUND: We have previously shown that infants who were extremely low birth weight (ELBW) are particularly vulnerable to problems related to inattention and hyperactivity at school age. It is not known whether these problems persist to adolescence. OBJECTIVE: To explore and compare the levels of psychopathology in a regional cohort of ELBW infants and sociodemographically matched term controls as reported by teens and their parents. DESIGN/METHODS: Cross-sectional cohort study/geographically defined region. PARTICIPANTS: TEENS: ELBW 141/169 (83%) and control 122/145 (84%), aged 12 to 16 years. PARENTS: ELBW 143/169 (85%) and control 123/145 (85%). Both cohorts and their parents completed the Ontario Child Health Study-Revised questionnaire with 6 behavioral subsca1es: conduct disorder, oppositional defiant disorder, attention-deficit/hyperactivity disorder (ADHD), overanxious, separation anxiety, and depression. RESULTS: By teen self-report, there were no significant differences between ELBW and control teens on any of the 6 subscale scores. However, parents of ELBW teens reported significantly higher scores than parents of control for depression and ADHD based on 2-way analysis of variance (group x gender). Comparison within teen/parent dyads showed that both cohorts of teens reported significantly higher scores than their parents. Multivariable analyses on behavioral subscale scores demonstrated a number of variables that were significant predictors by parent report: group (ELBW vs control), gender, family function, developmental quotient, maternal mood, and socioeconomic status; no predictors were significant by teen report. There were no statistically significant interaction effects for any of the models. These models explained a greater percent of the variance in behavioral scores for parents than for teens (12.5%-22.0% vs 3.4%-8.2%). Results were similar when teens with neurosensory impairment were excluded. CONCLUSIONS: This study is unique in the inclusion of both parent and teen self-report of behaviors. Significant differences were apparent only by parent report for ADHD and depression among ELBW teens. Significant predictors of behavioral scores for parents include group, gender, family function, developmental quotient, maternal mood, and socioeconomic status.

Self-esteem of adolescents who were born prematurely.

OBJECTIVE: To determine whether there are any differences between the self-esteem of a cohort of adolescents who were extremely low birth weight (ELBW) in comparison with term controls (Cs); and to test the associations between self-esteem and several predefined predictor variables. BACKGROUND: Self-esteem is considered to play a significant role in psychological adjustment and scholastic success. Little information exists on how adolescents who were ELBW regard themselves. DESIGN/METHODS: Longitudinal follow-up of a regional cohort of 132/169 (78%) ELBW survivors and 127/145 (88%) sociodemographically matched Cs, born between 1977 and 1982. MEASURES: Harter Adolescent Self-Perception Profile (1988) with 9 dimensions, including Global Self-Worth, socioeconomic status (Hollingshead), height-for-age and weight-for-age z scores, and Wide Range Achievement Test---Revised (WRAT-R; Reading, Spelling, and Arithmetic). DATA ANALYSIS: General linear model multiple analyses of covariance were performed to determine whether significant relationships existed between the 9 self-esteem dimensions and the independent variables of birth weight status and gender, and the covariates of age, socioeconomic status, physical development, and academic achievement. RESULTS: Global Self Worth was similar for ELBW and Cs (means: 3.1 and 3.2). Multivariate effects revealed no interactions, but significant main effects emerged for birth weight status, gender, weight-for-age z scores, age in months, and for all 3 WRAT-R subtests, all effect sizes medium to large. Follow-up analysis of covariance revealed medium-size gender effects for athletic competence (means: 3.1 and 2.6), and physical appearance (means: 2.9 and 2.5), where boys rated themselves significantly higher on both domains; and age effects, where older teens rated themselves better for job competence. Significant but small effect sizes emerged for the following: 1) weight-for-age z scores, where heavier youth rated themselves higher on close friendships, 2) gender, where girls had higher ratings for close friendships, 3) birth weight, where Cs rated themselves higher on athletic competence, and 4) WRAT-R math effect, where children with higher math scores rated themselves better on scholastic competence. CONCLUSIONS: Overall, ELBW teenagers do not differ significantly from C teenagers on most dimensions of self-esteem. Gender effects emerged on some Harter domains.