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自闭症与源自母亲的15号染色体长臂畸变

Autism and maternally derived aberrations of chromosome 15q.

作者信息

Schroer R J, Phelan M C, Michaelis R C, Crawford E C, Skinner S A, Cuccaro M, Simensen R J, Bishop J, Skinner C, Fender D, Stevenson R E

机构信息

Greenwood Genetic Center, South Carolina 29646, USA.

出版信息

Am J Med Genet. 1998 Apr 1;76(4):327-36. doi: 10.1002/(sici)1096-8628(19980401)76:4<327::aid-ajmg8>3.0.co;2-m.

Abstract

Of the chronic mental disabilities of childhood, autism is causally least well understood. The former view that autism was rooted in exposure to humorless and perfectionistic parenting has given way to the notion that genetic influences are dominant underlying factors. Still, identification of specific heritable factors has been slow with causes identified in only a few cases in unselected series. A broad search for genetic and environmental influences that cause or predispose to autism is the major thrust of the South Carolina Autism Project. Among the first 100 cases enrolled in the project, abnormalities of chromosome 15 have emerged as the single most common cause. The four abnormalities identified include deletions and duplications of proximal 15q. Other chromosome aberrations seen in single cases include a balanced 13;16 translocation, a pericentric inversion 12, a deletion of 20p, and a ring 7. Candidate genes involved in the 15q region affected by duplication and deletion include the ubiquitin-protein ligase (UBE3A) gene responsible for Angelman syndrome and genes for three GABA(A) receptor subunits. In all cases, the deletions or duplications occurred on the chromosome inherited from the mother.

摘要

在儿童慢性精神残疾中,自闭症的病因是最不清楚的。以前认为自闭症源于接触缺乏幽默感和完美主义的养育方式的观点,已被遗传影响是主要潜在因素的观念所取代。然而,特定遗传因素的识别进展缓慢,在未经过挑选的病例系列中,仅有少数病例确定了病因。对导致或易患自闭症的遗传和环境影响进行广泛研究,是南卡罗来纳州自闭症项目的主要方向。在该项目登记的首批100例病例中,15号染色体异常已成为最常见的单一病因。已确定的四种异常包括近端15q的缺失和重复。在个别病例中发现的其他染色体畸变包括13;16平衡易位、12号染色体臂间倒位、20p缺失和7号环状染色体。受重复和缺失影响的15q区域中涉及的候选基因包括与天使综合征相关的泛素蛋白连接酶(UBE3A)基因以及三个GABA(A)受体亚基的基因。在所有病例中,缺失或重复都发生在从母亲遗传来的染色体上。

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