Journal article
Agreement between parents' and clinical researchers' ratings of behavioral problems in children with fragile X syndrome and chromosome 15 imprinting disorders
M Arpone, L Bretherton, DJ Amor, SJC Hearps, C Rogers, MJ Field, MF Hunter, L Santa Maria, AM Alliende, J Slee, DE Godler, EK Baker
Research in Developmental Disabilities | PERGAMON-ELSEVIER SCIENCE LTD | Published : 2022
Abstract
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 Aber..
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Awarded by Foundation for Prader-Willi Research
Funding Acknowledgements
This study was supported by the Victorian Government's Operational Infrastructure Support Program, with the salaries supported by NHMRC project grants (1049299, 1103389 to D.E.G); Murdoch Children's Research Institute, Royal Children's Hospital Foundation (D.E.G.); Next Generation Clinical Researchers Program-Career Development Fellowship, funded by the Medical Research Future Fund (MRF1141334 to D.E.G.); the Financial Markets Foundation for Children (Australia) (no. 2017 - 361 to D.E.G. and D.J.A.); the Foundation for Angelman Syndrome Therapeutics (FAST; Australia to E.K.B.); and the Foundation for Prader-Willi Research (FPWR to D.E.G., D.J.A and E.K.B.). M.J.F. and C.R. were supported by the Genetics of Learning Disability (GOLD) Service. M.A. was supported by an Australian Postgraduate Award, the International Postgraduate Research Scholarships (IPRS) and the Research Training Program Fee offset scholarship awarded by the University of Melbourne, and in part by the Diagnosis and Development group of the Murdoch Children's Research Institute.