Aim We examined key features of two outcome measures for social dysfunction and autism spectrum disorder traits, the Social Responsiveness Scale, Second Edition (SRS‐2) and the Social Skills Improvement System – Rating Scales (SSIS‐RS), in children with neurofibromatosis type 1 (NF1). The aim of the study was to provide objective evidence as to which behavioural endpoint should be used in clinical trials. Method Cross‐sectional behavioural and demographic data were pooled from four paediatric NF1 tertiary referral centres in Australia and the United States (N=122; 65 males, 57 females; mean age [SD] 9y 2mo [3y], range 3–15y). Results Distributions of SRS‐2 and SSIS‐RS scores were unimodal and both yielded deficits, with a higher proportion of severely impaired scores on the SRS‐2 (16.4%) compared to the SSIS‐RS (8.2%). Pearson’s product‐moment correlations revealed that both questionnaires were highly related to each other (r=−0.72, p<0.001) and to measures of adaptive social functioning (both p<0.001). Both questionnaires were significantly related to attention‐deficit/hyperactivity disorder symptoms, but only very weakly associated with intelligence. Interpretation The SRS‐2 and SSIS‐RS capture social dysfunction associated with NF1, suggesting both may be suitable choices for assessing social outcomes in this population in a clinical trial. However, careful thought needs to be given to the nature of the intervention when selecting either as a primary endpoint. What this paper adds The Social Responsiveness Scale, Second Edition yielded a large deficit relative to population norms. The Social Skills Improvement System – Rating Scales yielded a moderate deficit relative to population norms. Both scales were highly correlated, suggesting that they are measuring a unitary construct.

Objective: Social skills difficulties are commonly reported by parents and teachers of school age (SA) children with neurofibromatosis type 1 (NF1). Investigations of social skills of young children with NF1 are scarce. This study aimed to characterize the emergence of social skills challenges beginning in early childhood, examine social skills longitudinally into SA, and explore interrelations with attention-deficit hyperactivity disorder (ADHD) symptomatology and cognitive functioning among children with NF1 crosssectionally and longitudinally. Method: Three samples of children with NF1 and their parents participated: (1) early childhood (n 5 50; ages 3-6; mean [M] 5 3.96, SD 5 1.05), ( 2) SA (n 5 40; ages 9-13; [M] 5 10.90, SD 5 1.59), and (3) both early childhood and SA (n 5 25). Parent-reported social skills (Social Skills Rating System and Social Skills Improvement System), ADHD symptomatology (Conners Parent Rating Scales -Revised and Conners -Third Edition), and parent-reported cognitive abilities (Differential Ability Scales -Second Edition) were evaluated. Results: Parental ratings of social skills were relatively stable throughout childhood. Ratings of social skills at the end of early childhood significantly predicted school-age social skills. Parental ratings of ADHD symptomatology showed significant negative relations with social skills. Early childhood inattentive symptoms predicted school-age social skills ratings. Cognitive functioning was not significantly related to social skills. Conclusion: Parent-reported social skills difficulties are evident during early childhood. This work adds to the literature by describing the frequency and stability of social skills challenges in early childhood and in the school-age period in NF1. Research about interventions to support social skills when difficulties are present is needed.

Objective: Children with neurofibromatosis type 1 (NF1) demonstrate poorer adaptive functioning compared with same-aged peers; however, there is limited research about the longitudinal pattern of adaptive behavior. The aim of this investigation was to examine parent-reported adaptive behavior of children with NF1 longitudinally beginning in early childhood and to examine relations with executive functioning. Method: Children with NF1 were assessed during early childhood (n = 59; aged 3–7; mean = 4.8, SD = 1.42) or school age (n = 39; aged 9–13; mean = 10.85, SD = 1.58), and a subset was seen at both time points (n = 26). The Scales of Independent Behavior—Revised was used to assess adaptive functioning, and the Behavior Rating Inventory of Executive Function—Preschool Version/Behavior Rating Inventory of Executive Function was used to evaluate everyday executive functioning. Results: Adaptive behavior in early childhood was significantly correlated with adaptive behavior at school age (with the exception of social interaction and communication skills) and was significantly poorer at school age. The frequency of difficulties increased over time for overall adaptive behavior and motor skills. Executive functioning was related to adaptive behavior cross-sectionally within early childhood and at school age and showed longitudinal predictive value over time. Conclusion: This research contributes to the limited NF1 adaptive behavior literature by characterizing the longitudinal pattern of adaptive behavior and relations with executive abilities.

The COVID-19 pandemic sparked a worldwide transition to providing online services overnight, highlighting the urgent need for empirically supported telehealth interventions. The current study examined the effects of PEERS® for Adolescents Telehealth, an adaptation from the original social skills intervention developed for in-person provision, among 22 autistic adolescents and their caregivers. To evaluate the intervention, caregivers completed questionnaires assessing core autistic features and frequency of get-togethers. Adolescents completed questionnaires measuring social knowledge and frequency of get-togethers. Improvements in social skills knowledge, increased get-togethers, and decreased core autistic symptoms were evident. Preliminary results suggest PEERS® for Adolescents Telehealth improves social competence, as found for the in-person version. Further research exploring the equivalence of telehealth to in-person social skills intervention is recommended.

Children with neurofibromatosis type 1 (NF1) often demonstrate difficulties with attention and executive functioning that can be evident starting at a young age. There has been little research about which measures of attention are most suitable for use with young children with NF1. This pilot study explored several computerized measures of attention, a digits forward task, and parent report measures of attention to compare their reliability, validity, and the degree to which they capture attention difficulty in this population. Participants with NF1 ages 4 to 6 years were seen for one ( n= 2) or two ( n= 18) time points. Statistical analyses for evaluating evidence for test-retest reliability, convergent and discriminant validity, practice effects, and identification of difficulties were conducted. Each measure demonstrated relative strengths and weaknesses, and there may not be a “one size fits all” measure for use with young children with NF1. However, the Behavior Rating Inventory of Executive Function Preschool/Second Edition, Conners Early Childhood Inattention/Hyperactivity Scale, and the Conners Kiddie Continuous Performance Test Second Edition generally had the highest reliability and most evidence of validity. More specific recommendations are provided for the appropriate measure to use in clinical and research batteries.