Distinctive patterns of memory function in subgroups of females with Turner syndrome: evidence for imprinted loci on the X-chromosome affecting neurodevelopment

被引:71
作者
Bishop, DVM
Canning, E
Elgar, K
Morris, E
Jacobs, PA
Skuse, DH
机构
[1] Univ Oxford, Dept Expt Psychol, Oxford OX1 3UD, England
[2] Inst Child Hlth, Behav Sci Unit, London, England
[3] Salisbury District Hosp, Wessex Human Genet Inst, Salisbury, Wilts, England
基金
英国惠康基金;
关键词
imprinting; verbal; visuospatial; monosomy; brain; lateralization;
D O I
10.1016/S0028-3932(99)00118-9
中图分类号
B84 [心理学]; C [社会科学总论]; Q98 [人类学];
学科分类号
03 ; 0303 ; 030303 ; 04 ; 0402 ;
摘要
X-monosomy is a form of Turner syndrome (TS) in which an entire X chromosome is missing. It is usually assumed that neuropsychological deficits in females with TS result from insufficient dosage of gene products from alleles on the sex chromosomes. If so, then parental origin of the single X chromosome should be immaterial. However, if there are imprinted genes on the X chromosome affecting brain development, neuropsychological development will depend on the parental origin of the single X chromosome. We contrasted verbal and visuospatial memory in females with a single paternal X chromosome (45,X-p) and those with a single maternal X (45,X-m). Neither group showed ally impairment on immediate story recall; if anything, performances was above control levels. Groups did not differ on a measure of delayed recall. However, when delayed recall was considered after adjusting for level of immediate recall, 45,X-m females showed enhanced verbal forgetting relative to controls over a delay. On the Ray figure, both groups were poor at copying the figure, but, after adjusting scores for initial copy score and strategy, only the 45,X-p,XP females showed disproportionate forgetting relative to controls. We propose there may be one or mere imprinted genes on the X chromosome that affect the development of lateralised brain regions important for memory function. (C) 2000 Elsevier Science Ltd. All rights reserved.
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页码:712 / 721
页数:10
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