Young adult male carriers of the fragile X premutation exhibit genetically modulated impairments in visuospatial tasks controlled for psychomotor speed.
Young adult male carriers of the fragile X premutation exhibit genetically modulated impairments in visuospatial tasks controlled for psychomotor speed.
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DOI：
10.1186/1866-1955-4-26
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发表时间：
2012-11-13
影响因子：
4.9
通讯作者：
Simon TJ
中科院分区：
文献类型：
--
作者：
Wong LM;Goodrich-Hunsaker NJ;McLennan Y;Tassone F;Harvey D;Rivera SM;Simon TJ
A previous study reported enhanced psychomotor speed, and subtle but significant cognitive impairments, modulated by age and by mutations in the fragile X mental retardation 1 (FMR1) gene in adult female fragile X premutation carriers (fXPCs). Because male carriers, unlike females, do not have a second, unaffected FMR1 allele, male fXPCs should exhibit similar, if not worse, impairments. Understanding male fXPCs is of particular significance because of their increased risk of developing fragile X-associated tremor/ataxia syndrome (FXTAS). Male fXPCs (n = 18) and healthy control (HC) adults (n = 26) aged less than 45 years performed two psychomotor speed tasks (manual and oral) and two visuospatial tasks (magnitude comparison and enumeration). In the magnitude comparison task, participants were asked to compare and judge which of two bars was larger. In the enumeration task, participants were shown between one and eight green bars in the center of the screen, and asked to state the total number displayed. Enumeration typically proceeds in one of two modes: subitizing, a fast and accurate process that works only with a small set of items, and counting, which requires accurate serial-object detection and individuation during visual search. We examined the associations between the performance on all tasks and the age, full-scale intelligent quotient, and CGG repeat length of participants. We found that in the magnitude comparison and enumeration tasks, male fXPCs exhibited slower reaction times relative to HCs, even after controlling for simple reaction time. Our results indicate that male fXPCs as a group show impairments (slower reaction times) in numerical visuospatial tasks, which are consistent with previous findings. This adds to a growing body of literature characterizing the phenotype in fXPCs who are asymptomatic for FXTAS. Future longitudinal studies are needed to determine how these impairments relate to risk of developing FXTAS.
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影响因子：
2.9
作者：
Goodrich-Hunsaker, Naomi J;Wong, Ling M;Simon, Tony J
通讯作者：
Simon, Tony J
DOI：
10.4088/jcp.08m04476
发表时间：
2009-06
期刊：
The Journal of clinical psychiatry
影响因子：
--
作者：
Bourgeois JA;Coffey SM;Rivera SM;Hessl D;Gane LW;Tassone F;Greco C;Finucane B;Nelson L;Berry-Kravis E;Grigsby J;Hagerman PJ;Hagerman RJ
通讯作者：
Hagerman RJ
影响因子：
2.7
作者：
Hunsaker, Michael R.;Goodrich-Hunsaker, Naomi J.;Berman, Robert F.
通讯作者：
Berman, Robert F.
影响因子：
9.9
作者：
Cornish, Kim M.;Hocking, Darren R.;Kogan, Cary S.
通讯作者：
Kogan, Cary S.
影响因子：
2.4
作者：
Allen, Emily Graves;Hunter, Jessica E.;Sherman, Stephanie L.
通讯作者：
Sherman, Stephanie L.