Measurement considerations in pediatric research on autism spectrum disorders

Walsh, Karin S.; Rau, Srishti

doi:10.1016/bs.pbr.2018.09.015

Cited by 1 publication

(2 citation statements)

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“…Concerns within the social domain are widely observed across RASopathies, and clinical assessments indicate a higher than expected prevalence of autism spectrum disorder (ASD) symptomatology and diagnosis [ 6 , 7 , 14 – 17 ]. The nature of social behavior in RASopathies relative to ASD, a neurodevelopmental disorder defined by behavioral criteria, is nevertheless poorly understood [ 18 ]. Diagnosis of ASD requires the presence of impairments in social communication and interaction as well as restricted or repetitive activities, interests, or behaviors [ 19 ].…”

Section: Introductionmentioning

confidence: 99%

“…Estimates of ASD prevalence among individuals with RASopathies have varied widely across studies. These estimates appear to depend heavily upon the specific assessment tools and methodology used as well as study sample characteristics [ 18 ]. Among clinically referred cohorts, ASD estimates in RASopathies are generally higher as compared to epidemiological samples [ 16 ].…”

Section: Introductionmentioning

confidence: 99%

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Social behavior in RASopathies and idiopathic autism

Foy

Hudock

Shanley

et al. 2022

J Neurodevelop Disord

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Background RASopathies are genetic syndromes that result from pathogenic variants in the RAS-MAPK cellular signaling pathway. These syndromes, which include neurofibromatosis type 1, Noonan syndrome, cardiofaciocutaneous syndrome, and Costello syndrome, are associated with a complex array of medical and behavioral health complications. Despite a heightened risk for social challenges and autism spectrum disorder (ASD), few studies have compared different aspects of social behavior across these conditions. It is also unknown whether the underlying neuropsychological characteristics that contribute to social competence and socially empathetic (“prosocial”) behaviors differ in children with RASopathies as compared to children with nonsyndromic (i.e., idiopathic) ASD. Methods In this cross-sectional, survey-based investigation, caregivers of preschool and school-aged children with RASopathies (n = 202) or with idiopathic ASD (n = 109) provided demographic, medical, and developmental information about their child, including psychiatric comorbidities. For children who were able to communicate verbally, caregivers also completed standardized rating scales to assess social competence and empathetic behavior as well as symptoms of hyperactivity/inattention and emotional problems. Results As compared to children with idiopathic ASD, children with RASopathies were rated as demonstrating more resilience in the domain of empathy relative to their overall social competence. Similarities and differences emerged in the psychological factors that predicted social behavior in these two groups. Stronger communication skills and fewer hyperactive-impulsive behaviors were associated with increased empathy and social competence for both groups. Greater emotional challenges were associated with lower social competence for children with RASopathies and stronger empathy for children with idiopathic ASD. Among children with RASopathy and a co-occurring ASD diagnosis, socially empathetic behaviors were observed more often as compared to children with idiopathic ASD. Conclusions Findings suggest that the development of social behavior among children with RASopathies involves a distinct pattern of strengths and weaknesses as compared to a behaviorally defined disorder (idiopathic ASD). Identification of areas of resilience as well as behavioral and social challenges will support more targeted intervention.

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