Srinivasan, R;
Wolstencroft, J;
Erwood, M;
Raymond, FL;
van den Bree, M;
Hall, J;
Skuse, D;
(2019)
Mental health and behavioural problems in children with XXYY: a comparison with intellectual disabilities.
Journal of Intellectual Disability Research
, 63
(5)
pp. 477-488.
10.1111/jir.12607.
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Abstract
Background The phenotype of children with XXYY has predominantly been defined by comparison to other sex chromosome aneuploidies trisomies affecting male children; however, the intellectual ability of children with XXYY is lower than children with other sex chromosome aneuploidies trisomies. It is not known to what extent the phenotype identified to date is specific to XXYY, rather than a reflection of lower IQ. This study evaluates the mental health and behaviour of children with XXYY, in comparison to children with intellectual disabilities of heterogeneous genetic origin. Methods Fifteen children with XXYY and 30 controls matched for age (4–14 years), sex and intellectual ability were ascertained from the IMAGINE ID study. IMAGINE ID participants have intellectual disabilities due to genetic anomalies confirmed by National Health Service Regional Genetic Centre laboratories. The mental health and behaviour of participants was examined with the Development and Well‐being Assessment and the Strengths and Difficulties Questionnaire. Results Children with XXYY experienced significantly more frequent and intense temper outbursts than the control group. Conclusion Our results suggest that temper outbursts may be specifically associated with the XXYY phenotype. These problems have a significant impact on the daily lives of boys with XXYY and their families. It is crucial to ensure that families are well supported to manage these difficulties.
| Type: | Article |
|---|---|
| Title: | Mental health and behavioural problems in children with XXYY: a comparison with intellectual disabilities |
| Open access status: | An open access version is available from UCL Discovery |
| DOI: | 10.1111/jir.12607 |
| Publisher version: | https://doi.org/10.1111/jir.12607 |
| Language: | English |
| Additional information: | This version is the author accepted manuscript. For information on re-use, please refer to the publisher’s terms and conditions. |
| Keywords: | Social Sciences, Science & Technology, Life Sciences & Biomedicine, Education, Special, Genetics & Heredity, Clinical Neurology, Psychiatry, Rehabilitation, Education & Educational Research, Neurosciences & Neurology, behaviour, conduct, genetics, mental health, sex chromosomes, SEX-CHROMOSOME ANEUPLOIDY, DEFICIT HYPERACTIVITY DISORDER, OPPOSITIONAL DEFIANT DISORDER, PAST 10 YEARS, CONDUCT DISORDER, ADOLESCENTS, XYY, QUESTIONNAIRE, STRENGTHS, 48,XXXY |
| UCL classification: | UCL UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Brain Sciences UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Brain Sciences > Division of Psychiatry UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Population Health Sciences > UCL GOS Institute of Child Health UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Population Health Sciences > UCL GOS Institute of Child Health > Population, Policy and Practice Dept |
| URI: | https://discovery.ucl.ac.uk/id/eprint/10075429 |
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