作者
Marta Arpone, Lesley Bretherton, David J Amor, Stephen JC Hearps, Carolyn Rogers, Michael J Field, Matthew F Hunter, Lorena Santa Maria, Angelica M Alliende, Jennie Slee, David E Godler, Emma K Baker
发表日期
2022/12/1
期刊
Research in Developmental Disabilities
卷号
131
页码范围
104338
出版商
Pergamon
简介
Background
Despite the increasing number of clinical trials involving children with neurodevelopmental disorders, appropriate and objective outcome measures for behavioral symptoms are still required.
Aim
This study assessed the agreement between parents' and clinical researchers' ratings of behavioral problem severity in children with fragile X syndrome (FXS) and chromosome 15 imprinting disorders.
Methods and Procedures
The cohort comprised 123 children (64% males), aged 3–17 years, with FXS (n = 79), Prader-Willi (PWS; n = 19), Angelman (AS; n = 15), and Chromosome 15q duplication (n = 10) syndromes. Specific items from the Autism Diagnostic Observation Schedule-Second Edition and Aberrant Behavior Checklist-Community Edition mapping to corresponding behavioral domains were selected ad-hoc, to assess behavioral problems.
Outcomes and Results
Inter-rater agreement for the cohort was …
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