Suppr超能文献

鉴定 ANKRD11 和 ZNF778 为新的 16q24.3 微缺失综合征相关自闭症和可变认知障碍的候选基因。

Identification of ANKRD11 and ZNF778 as candidate genes for autism and variable cognitive impairment in the novel 16q24.3 microdeletion syndrome.

机构信息

Department of Human Genetics, Radboud University Nijmegen Medical Centre, PO Box 9101, Nijmegen 6500 HB, The Netherlands.

出版信息

Eur J Hum Genet. 2010 Apr;18(4):429-35. doi: 10.1038/ejhg.2009.192. Epub 2009 Nov 18.

Abstract

The clinical use of array comparative genomic hybridization in the evaluation of patients with multiple congenital anomalies and/or mental retardation has recently led to the discovery of a number of novel microdeletion and microduplication syndromes. We present four male patients with overlapping molecularly defined de novo microdeletions of 16q24.3. The clinical features observed in these patients include facial dysmorphisms comprising prominent forehead, large ears, smooth philtrum, pointed chin and wide mouth, variable cognitive impairment, autism spectrum disorder, structural anomalies of the brain, seizures and neonatal thrombocytopenia. Although deletions vary in size, the common region of overlap is only 90 kb and comprises two known genes, Ankyrin Repeat Domain 11 (ANKRD11) (MIM 611192) and Zinc Finger 778 (ZNF778), and is located approximately 10 kb distally to Cadherin 15 (CDH15) (MIM 114019). This region is not found as a copy number variation in controls. We propose that these patients represent a novel and distinctive microdeletion syndrome, characterized by autism spectrum disorder, variable cognitive impairment, facial dysmorphisms and brain abnormalities. We suggest that haploinsufficiency of ANKRD11 and/or ZNF778 contribute to this phenotype and speculate that further investigation of non-deletion patients who have features suggestive of this 16q24.3 microdeletion syndrome might uncover other mutations in one or both of these genes.

摘要

患者存在多发先天畸形和/或智力障碍时,临床采用基因芯片比较基因组杂交技术,最近发现了一些新的微缺失和微重复综合征。本文报道 4 例男性患者,他们存在新发的、分子定义的、16q24.3 区域的不同程度的杂合性缺失。这些患者的临床表现包括:额部宽大、耳大、人中短、下颌尖、口裂宽等面部畸形,认知障碍、自闭症谱系障碍、脑结构异常、癫痫发作和新生儿血小板减少症。虽然缺失的大小不同,但重叠的共同区域仅有 90kb,包含两个已知基因:ANKRD11(MIM 611192)和 Zinc Finger 778(ZNF778),位于 Cadherin 15(CDH15)(MIM 114019)下游约 10kb 处。该区域在对照组中未发现拷贝数变异。我们提出这些患者代表了一种新的、独特的微缺失综合征,其特征为自闭症谱系障碍、认知障碍、面部畸形和脑异常。我们推测 ANKRD11 和/或 ZNF778 的杂合性缺失导致了该表型,并推测对具有该 16q24.3 微缺失综合征特征的非缺失患者进行进一步研究,可能会发现这两个基因中的一个或两个基因的其他突变。

相似文献

引用本文的文献

本文引用的文献

4
FOXG1 is responsible for the congenital variant of Rett syndrome.FOXG1基因与瑞特综合征的先天性变异有关。
Am J Hum Genet. 2008 Jul;83(1):89-93. doi: 10.1016/j.ajhg.2008.05.015. Epub 2008 Jun 19.
7
The epigenetic origins of mental retardation.智力迟钝的表观遗传起源。
Clin Genet. 2008 Jun;73(6):528-30. doi: 10.1111/j.1399-0004.2008.00979_1.x. Epub 2008 Apr 22.
9
Structural variation of chromosomes in autism spectrum disorder.自闭症谱系障碍中染色体的结构变异
Am J Hum Genet. 2008 Feb;82(2):477-88. doi: 10.1016/j.ajhg.2007.12.009. Epub 2008 Jan 17.

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验