caa a22 4500 605476683 CHVBK 20210128100357.0 cr unu---uuuuu 210128e20150901xx s 000 0 eng 10.1007/s00787-014-0662-4 doi (NATIONALLICENCE)springer-10.1007/s00787-014-0662-4 Behavior in children with Prader-Willi syndrome before and during growth hormone treatment: a randomized controlled trial and 8-year longitudinal study [Elektronische Daten] [Sin Lo, Elbrich Siemensma, Dederieke Festen, Philippe Collin, Anita Hokken-Koelega] Information on behavior of children with Prader-Willi syndrome (PWS) and the effect of growth hormone (GH) treatment is scarce. Parents report less problem behavior during GH treatment. Forty-two pre-pubertal children, aged 3.5-14years were studied in a randomized controlled GH trial (RCT) during 2 years, followed by a longitudinal study during 8 years of GH treatment. Behavior was measured annually by the Developmental Behavior Checklist for children with intellectual disability (DBC) and a Dutch questionnaire to evaluate social behavioral problems in children, the Children's Social Behavior Questionnaire (CSBQ). Problem behavior measured by the DBC in children with PWS was similar compared to peers with comparable intellectual disability. Scores on ‘Social disabilities' subscale were however significantly higher compared to the DBC total score (p<0.01). A lower IQ was associated with more self-absorbed behavior, more communication problems and more problem behavior in general. Problem behavior measured by the CSBQ was similar compared to peers with a comparable intellectual disability, but children with PWS scored significantly higher on the ‘Not tuned', ‘Understanding', and ‘Stereotyped' subscales than the CSBQ total score (p<0.05 for all subscales and p=0.001 for the ‘Not tuned'-subscale). There were no significant effects of GH treatment during the RCT and 8years of GH treatment. Children with PWS showed similar problem behavior as a reference population with a comparable intellectual disability. Social problems were the most pronounced within-problem behavior in PWS. In contrast to our expectations and parents reports, our study shows no improvement but also no deterioration of behavioral problems in children with PWS during long-term GH treatment. Springer-Verlag Berlin Heidelberg, 2014 Prader-Willi syndrome nationallicence Behavior nationallicence Growth hormone nationallicence Lo Sin Dutch Growth Research Foundation, Westzeedijk 106, 3016 AH, Rotterdam, The Netherlands aut Siemensma Elbrich Dutch Growth Research Foundation, Westzeedijk 106, 3016 AH, Rotterdam, The Netherlands aut Festen Dederieke Dutch Growth Research Foundation, Westzeedijk 106, 3016 AH, Rotterdam, The Netherlands aut Collin Philippe Gastenhof, Koraalgroep/de Hondsberg, Stationsstraat 10, 6131 AZ, Sittard, The Netherlands aut Hokken-Koelega Anita Dutch Growth Research Foundation, Westzeedijk 106, 3016 AH, Rotterdam, The Netherlands aut European Child & Adolescent Psychiatry Springer Berlin Heidelberg 24/9(2015-09-01), 1091-1101 1018-8827 24:9<1091 2015 24 787 https://doi.org/10.1007/s00787-014-0662-4 text/html Onlinezugriff via DOI BK010053 XK010053 XK010000 Metadata rights reserved Springer special CC-BY-NC licence nationallicence 1 research-article jats NATIONALLICENCE NATIONALLICENCE NL-springer NATIONALLICENCE 856 40 https://doi.org/10.1007/s00787-014-0662-4 text/html Onlinezugriff via DOI NATIONALLICENCE 700 1- Lo Sin Dutch Growth Research Foundation, Westzeedijk 106, 3016 AH, Rotterdam, The Netherlands aut NATIONALLICENCE 700 1- Siemensma Elbrich Dutch Growth Research Foundation, Westzeedijk 106, 3016 AH, Rotterdam, The Netherlands aut NATIONALLICENCE 700 1- Festen Dederieke Dutch Growth Research Foundation, Westzeedijk 106, 3016 AH, Rotterdam, The Netherlands aut NATIONALLICENCE 700 1- Collin Philippe Gastenhof, Koraalgroep/de Hondsberg, Stationsstraat 10, 6131 AZ, Sittard, The Netherlands aut NATIONALLICENCE 700 1- Hokken-Koelega Anita Dutch Growth Research Foundation, Westzeedijk 106, 3016 AH, Rotterdam, The Netherlands aut NATIONALLICENCE 773 0- European Child & Adolescent Psychiatry Springer Berlin Heidelberg 24/9(2015-09-01), 1091-1101 1018-8827 24:9<1091 2015 24 787