Faculty Collaboration Database

Social skills and autism spectrum disorder symptoms in children with neurofibromatosis type 1: evidence for clinical trial outcomes. Dev Med Child Neurol 2020 Jul;62(7):813-819

Date

03/18/2020

Pubmed ID

32181506

DOI

10.1111/dmcn.14517

Scopus ID

2-s2.0-85081725703 (requires institutional sign-in at Scopus site)   17 Citations

Abstract

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.

Author List

Payne JM, Walsh KS, Pride NA, Haebich KM, Maier A, Chisholm A, Glad DM, Casnar CL, Rouel M, Lorenzo J, Del Castillo A, North KN, Klein-Tasman B

Authors

Danielle M. Glad PhD Instructor in the Neurology department at Medical College of Wisconsin
Bonita Klein-Tasman BA,MA,PhD Professor in the Psychology department at University of Wisconsin - Milwaukee

MESH terms used to index this publication - Major topics in bold

Adolescent
Autism Spectrum Disorder
Child
Child, Preschool
Clinical Trials as Topic
Cross-Sectional Studies
Female
Humans
Male
Neurofibromatosis 1
Psychiatric Status Rating Scales
Social Skills