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Executive dysfunction and the relation with behavioral problems in children with 47,XXY and 47,XXX

Genes, brain and behavior, 2015-02, Vol.14 (2), p.200-208 [Peer Reviewed Journal]

2015 John Wiley & Sons Ltd and International Behavioural and Neural Genetics Society ;2015 John Wiley & Sons Ltd and International Behavioural and Neural Genetics Society. ;ISSN: 1601-1848 ;EISSN: 1601-183X ;DOI: 10.1111/gbb.12203 ;PMID: 25684214 ;CODEN: GBBEAO

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  • Title:
    Executive dysfunction and the relation with behavioral problems in children with 47,XXY and 47,XXX
  • Author: van Rijn, S. ; Swaab, H.
  • Subjects: Aggression ; Behavior ; Behavioral problems ; Brain - physiopathology ; Child ; Children & youth ; Chromosomes ; Chromosomes, Human, X - genetics ; cognition ; Cognition Disorders - genetics ; Executive Function - physiology ; executive functions ; Female ; Formicidae ; Girls ; Humans ; Klinefelter syndrome ; Klinefelter Syndrome - genetics ; Male ; Neuropsychological Tests ; Problem Behavior - psychology ; Sex Chromosome Aberrations ; sex chromosome disorders ; Sex Chromosome Disorders of Sex Development - genetics ; Trisomy - genetics ; Trisomy X
  • Is Part Of: Genes, brain and behavior, 2015-02, Vol.14 (2), p.200-208
  • Description: Neuroimaging studies have shown that having an extra X chromosome is associated with abnormal structure and function of brain areas in the frontal lobe, which is crucially involved in executive functioning. However, there is little of knowledge of the type and severity of executive dysfunction, and the impact on emotional and behavioral problems. The present study aims to provide in this. In total, 40 children (23 boys with 47,XXY and 17 girls with 47,XXX) with an extra X chromosome and 100 non‐clinical controls (47 boys and 53 girls) participated in the study. The participants were 9–18 years old. Processing speed and executive functioning were assessed using the Amsterdam Neuropsychological Testbattery (ANT) and the Dysexecutive Questionnaire (DEX). Problems in emotional and behavioral functioning were assessed with the Childhood Behavior Checklist (CBCL). Children with an extra X chromosome showed deficits in inhibition, mental flexibility, sustained attention and visual working memory. Parental report showed high levels of everyday manifestations of executive dysfunction. More severe inhibition difficulties were associated with higher levels of thought problems, aggression and rule breaking behavior. Boys and girls with an extra X chromosome could not be differentiated based on severity of executive dysfunction, however, girls had lower information processing speed than boys. These findings suggest that executive dysfunction may be part of the phenotype of children with an extra X chromosome, impacting the ability to function adequately in everyday life. Furthermore, children with impairments in inhibition may have more problems in regulating their thinking, emotions and behavior. Neuroimaging studies have shown that having an extra X chromosome is associated with deviant structure and function of the frontal lobe, which is crucially involved in executive functioning. However, there is little of knowledge of executive dysfunction, and the impact on emotional and behavioral problems. Findings from this study suggests that executive dysfunction in areas of inhibition, mental flexibility, sustained attention and visual working memory may be part of the phenotype of both boys (47,XXY) and girls (47,XXX) with an extra X chromosome, impacting the ability to function adequately in everyday life. The finding that children with more impairments in inhibition had more problems in regulating their thinking and behavior, illustrates the relevance for emotional and behavioral problems.
  • Publisher: Oxford, UK: Blackwell Publishing Ltd
  • Language: English
  • Identifier: ISSN: 1601-1848
    EISSN: 1601-183X
    DOI: 10.1111/gbb.12203
    PMID: 25684214
    CODEN: GBBEAO
  • Source: Journals@Ovid Open Access Journal Collection Rolling
    MEDLINE
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