Martie L. Skinner

Autistic behavior in children with fragile X syndrome: Prevalence, stability, and the impact of FMRP

We examined autistic behavior in a cross‐sectional sample of 179 children with fragile X syndrome (FXS) and a longitudinal subset of 116 children using the Childhood Autism Rating Scale (CARS) to (a) determine a prevalence of autistic behavior in FXS, (b) examine the stability of autistic ratings over time, and (c) assess the association between the fragile X mental retardation protein (FMRP) and autistic behavior. Approximately 21% of the sample of 129 children (25.9% of boys) scored at or above the cutoff for autism. CARS scores increased slowly, yet significantly, over time, and low levels of FMRP were associated with higher mean levels of autistic behavior as measured by the CARS.

Persistent Effects of Early Childhood Education on High-Risk Children and Their Mothers

Child and mother outcomes are reported for the Abecedarian Project, an early childhood education, pediatric healthcare, and family support program for high-risk children and their mothers. Three randomized intervention conditions for at-risk participants were compared to a control condition. Randomized control group participants received family support social services, low-cost or free pediatric care, and child nutritional supplements but no additional educational program beyond what the parents and the local school system provided. The local community was generally affluent and well educated; disadvantaged families represented a small minority whose welfare was a high social and educational priority. The educational intervention conditions were (a) preschool education for the first 5 years of life plus a supplementary kindergarten through 2nd grade Educational Support Program (Preschool plus K-2 Support), (b) preschool education (Preschool Only), and (c) K-2 Educational Support Program (K-2 Only). Child outcomes were assessed with respect to cognitive development, academic achievement, grade retention, and special education placements. For mothers, the effect of having educational childcare during the preschool period was examined with respect to maternal educational gains and employment. Positive and systematic cognitive and academic achievement differences were found for children in the preschool treatment conditions, particularly when that condition was combined with the K-2 Educational Support Program. High-quality, consistently available preschool education was also associated with greater maternal educational advancement and higher levels of employment particularly for teenage mothers. These findings have direct relevance for education and welfare policies.

Autistic Behavior, FMR1 Protein, and Developmental Trajectories in Young Males with Fragile X Syndrome

In the context of a longitudinal study, we assessed the relationship between ratings of autistic behavior, FMR1 protein expression (FMRP), and the developmental trajectories of 55 young males with fragile X syndrome. Autistic behavior, as measured by the Childhood Autism Rating Scale, was not related to FMRP expression. However, autistic behavior was a significant predictor of both developmental status and developmental change. Boys with both autistic behavior and fragile X syndrome functioned at significantly lower levels of development and grew at significantly slower rates than those without autistic behavior. FMRP expression accounted for less variance in developmental level than did autistic behavior, and was not significantly related to slope (developmental change over time). No autistic behavior × FMRP interaction was found.

Early Development, Temperament, and Functional Impairment in Autism and Fragile X Syndrome.

We compared the developmental status, functional abilities, and temperament of 31 young boys with fragile X syndrome (FXS) who did not have autism, matched on chronological age, gender, and race, with 31 boys with autism but no FXS. Children with autism exhibited a more variable profile of development in comparison with a relatively flat profile for children with FXS. Children with autism were significantly more delayed in social skills and were rated by observers as exhibiting a greater degree of impairment in cognitive, communication, and social skills. On temperament ratings, both groups were slower to adapt, less persistent, and more withdrawing than the reference group. Boys with FXS were rated as more active than the referent group, whereas boys with autism were rated as less intense, more distractible, having a higher threshold for response, and less rhythmic than the reference group. A smaller three-group analysis compared boys with FXS, boys with autism, and boys with both FXS and autism. Children with both autism and FXS were substantially more delayed than children with autism or FXS alone.

Early developmental trajectories of males with fragile X syndrome.

Findings from a prospective longitudinal study of 46 boys with fragile X syndrome between the ages of 24 and 72 months were reported. Hierarchical linear modeling was used to construct and evaluate overall developmental trajectories and scores in five domains: Cognition, Communication, Adaptive, Motor, and Personal-Social. The children varied widely, with significant differences across individuals in both mean rate and level of performance. Overall development was significantly delayed, with a slope of .48--approximately half the rate expected for typically developing children. No differences were found in rates of growth across the five domains. Significant differences were found, however, in mean levels of performance. At every age tested, Motor and Adaptive scores were higher than Communication and Cognitive.

Needs and Supports Reported by Latino Families of Young Children with Developmental Disabilities.

We interviewed 200 Latino parents (50 Mexican couples, 50 Puerto Rican couples) living in the United States to determine needs and supports related to raising a child with a disability and to identify variables related to reported needs and supports. The pattern of needs expressed was similar to that found in previous studies, but the number was substantially higher. More support was reported from family and formal sources than from friends or informal sources. Using repeated measures of analysis of covariance involving six family variables and three child variables, we found that English language proficiency was the only variable to account for significant variance in needs and supports.

The effect of format modifications and reading comprehension on recall of informed consent information by low-income parents: a comparison of print, video, and computer-based presentations

A randomized trial comparing the amount of knowledge orally recalled from four different presentations of the same consent information was conducted in a non-clinic sample of 233 low-income parents who displayed a range of reading comprehension skill. The study simulated recruitment of children into one of two actual studies underway at another location: one involved high risk to participants, the other did not. Use of a non-clinic sample controlled for prior knowledge of the conditions, and avoiding discussion of the information further assured that differences in recalled information could be attributed more confidently to the format itself. The formats included the original written forms, enhanced print (simpler language, topic headings, pictures), narrated videotapes, and self-paced PowerPoint presentations via laptop computer with bulleted print information, pictures, and narration. No format-related differences in recalled information were found in the full sample but for the 124 individuals with reading comprehension scores at or below the 8th grade level, the enhanced print version tended to be more effective than either the original form or the video. Across all formats, more information was recalled about the low-risk study. The findings emphasize the necessity for clinicians and researchers to verify understanding of consent information, especially when there is risk of reduced literacy skill. Reliance on video to convey information in preference to well-done print media appeared questionable.

Self-injurious behavior in young boys with fragile X syndrome

In this study, we distributed surveys to 67 families of young boys with fragile X syndrome to determine the prevalence, onset, form, function, location, and correlates of self‐injurious behavior. Fifty‐five surveys were completed (82%). The mean age of the boys at the time of the survey was 80 months (range = 20–144). Self‐injurious behavior (SIB) was reported for 58% of the participants with a mean age of onset of 31 months. The mean number of forms of self‐injury was 2 per participant. Biting was the most commonly reported form of self‐injury with the fingers and back of the hand disproportionately targeted as the most prevalent self‐injury body site. There was no linear increase in risk of SIB with age past 25 months. SIB was reported as most likely to occur following the presentation of difficult task demands or changes in routine. Significant group differences were found between overall ratings of problem behavior for boys with self‐injury compared to those without self‐injury. Groups did not differ on measures of fragile X mental retardation protein (FMRP), autism status, adaptive behavior, or age first medicated. Results are discussed in terms of future research designed to further elucidate the behavioral phenotype of fragile X syndrome.

Setting Effects on Friendship Formation among Young Children with and without Disabilities

This study examined the effects of social setting on the friendship formation of 333 preschool children (120 children with disabilities and 213 typically developing children) enrolled in inclusive early childhood programs. The study found that typically developing children in specialized classrooms had significantly more friends than did children with disabilities in those same settings. In child care settings, however, the difference between the reported number of friendships for typically developing children and their peers with disabilities did not reach statistical significance. Implications are presented for considering how the social ecology of the early childhood classroom setting may influence social outcomes for young children enrolled in inclusive programs.

Role of Religion in the Lives of Latino Families of Young Children With Developmental Delays

We interviewed 250 parents of Mexican and Puerto Rican origin living in the United States who had young children with developmental delays to determine the role of religion in their lives. Quantitative results indicate that parents largely viewed themselves as religious, were affiliated with a formal religion, and participated in religious activities. Most parents viewed both church and faith as supportive, but faith was shown to provide more support. Repeated measures a analysis of variance found some intragroup variations in religious support and changes in support after learning of the childs condition. Thematic analysis revealed specific religious beliefs and practices parents viewed as supportive, and content and cultural models analyses indicated the religious frameworks by which parents interpreted their childs disability.