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.
Key Words
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Article info
Publication history
Published online: December 11, 2006
Accepted:
August 15,
2006
Received in revised form:
August 11,
2006
Received:
June 1,
2006
Identification
Copyright
© 2007 Society of Biological Psychiatry. Published by Elsevier Inc. All rights reserved.
