X-monosomy effects on visuospatial attention in mice: a candidate gene and implications for Turner syndrome and attention deficit hyperactivity disorder

Davies, William ORCID: https://orcid.org/0000-0002-7714-2440, Humby, Trevor ORCID: https://orcid.org/0000-0002-1840-1799, Isles, Anthony Roger ORCID: https://orcid.org/0000-0002-7587-5712, Burgoyne, Paul S. and Wilkinson, Lawrence Stephen ORCID: https://orcid.org/0000-0002-9337-6124 2007. X-monosomy effects on visuospatial attention in mice: a candidate gene and implications for Turner syndrome and attention deficit hyperactivity disorder. Biological psychiatry 61 (12) , pp. 1351-1360. 10.1016/j.biopsych.2006.08.011

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Abstract

BACKGROUND: The loss of all, or part of an X chromosome, in Turner syndrome (TS, 45,XO) results in deficits in attentional functioning. METHODS: Using a 39,XO mouse model, we tested the hypothesis that X-monosomy and/or parental origin of the single X chromosome may influence visuospatial attentional functioning in a 5-choice serial reaction time task (5-CSRTT). RESULTS: Under attentionally demanding conditions 39,XO mice displayed impaired discriminative response accuracy and slowed correct reaction times relative to 40,XX mice; these deficits were alleviated in a version of the task with reduced attentional demands. Parental origin of the X did not affect performance of the 5-CSRTT. In contrast, the attentional phenotype was rescued in 40,XY*X mice possessing a single maternally inherited X chromosome and a small Y*X chromosome that comprises a complete pseudoautosomal region (PAR), and a small X-specific segment. CONCLUSIONS: Our findings are consistent with an X-monosomy effect on attention and suggest the existence of X-linked gene(s) that escape X-inactivation, are present on the small Y*X chromosome and impact on attentional functioning; the strongest candidate gene is Sts, encoding steroid sulfatase. The data inform the TS literature and indicate novel genetic mechanisms that may be of general significance to the neurobiology of attention.

Item Type:	Article
Date Type:	Publication
Status:	Published
Schools:	Medicine Psychology MRC Centre for Neuropsychiatric Genetics and Genomics (CNGG) Neuroscience and Mental Health Research Institute (NMHRI)
Uncontrolled Keywords:	5-choice serial reaction time task; dehydroepiandrosterone sulfate; discriminative response accuracy; steroid sulfatase; X chromosome; Y*X chromosome
Publisher:	Elsevier
ISSN:	0006-3223
Last Modified:	28 Sep 2023 12:29
URI:	https://orca.cardiff.ac.uk/id/eprint/617

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