Frangiskakis J M, Ewart A K, Morris C A, Mervis C B, Bertrand J, Robinson B F, Klein B P, Ensing G J, Everett L A, Green E D, Pröschel C, Gutowski N J, Noble M, Atkinson D L, Odelberg S J, Keating M T
Department of Human Genetics, University of Utah Health Sciences Center, Salt Lake City 84112, USA.
Cell. 1996 Jul 12;86(1):59-69. doi: 10.1016/s0092-8674(00)80077-x.
To identify genes important for human cognitive development, we studied Williams syndrome (WS), a developmental disorder that includes poor visuospatial constructive cognition. Here we describe two families with a partial WS phenotype; affected members have the specific WS cognitive profile and vascular disease, but lack other WS features. Submicroscopic chromosome 7q11.23 deletions cosegregate with this phenotype in both families. DNA sequence analyses of the region affected by the smallest deletion (83.6 kb) revealed two genes, elastin (ELN) and LIM-kinase1 (LIMK1). The latter encodes a novel protein kinase with LIM domains and is strongly expressed in the brain. Because ELN mutations cause vascular disease but not cognitive abnormalities, these data implicate LIMK1 hemizygosity in imparied visuospatial constructive cognition.
为了识别对人类认知发展至关重要的基因,我们研究了威廉姆斯综合征(WS),这是一种发育障碍,包括视觉空间建构认知能力差。在这里,我们描述了两个具有部分WS表型的家族;受影响的成员具有特定的WS认知特征和血管疾病,但缺乏其他WS特征。亚显微染色体7q11.23缺失在两个家族中均与该表型共分离。对受最小缺失(83.6 kb)影响区域的DNA序列分析揭示了两个基因,即弹性蛋白(ELN)和LIM激酶1(LIMK1)。后者编码一种具有LIM结构域的新型蛋白激酶,并在大脑中强烈表达。由于ELN突变会导致血管疾病而非认知异常,这些数据表明LIMK1半合子状态与视觉空间建构认知受损有关。