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胼胝体发育不全、发育迟缓、自闭症谱系障碍、面部畸形以及与ZEB1基因缺失相关的后部多形性角膜营养不良。

Agenesis of the corpus callosum, developmental delay, autism spectrum disorder, facial dysmorphism, and posterior polymorphous corneal dystrophy associated with ZEB1 gene deletion.

作者信息

Chaudhry Ayeshah, Chung Brian H, Stavropoulos Dimitri J, Araya Marcela P, Ali Asim, Heon Elise, Chitayat David

机构信息

Department of Laboratory Medicine and Genetics, Trillium Health Partners, Mississauga, Ontario, Canada.

Department of Laboratory Medicine and Pathobiology, University of Toronto, Toronto, Ontario, Canada.

出版信息

Am J Med Genet A. 2017 Sep;173(9):2467-2471. doi: 10.1002/ajmg.a.38321. Epub 2017 Jul 25.

Abstract

We report on a girl diagnosed prenatally with agenesis of the corpus callosum (ACC) on fetal ultrasound and MRI. On postnatal follow-up she was noted to have developmental delay, facial dysmorphism, autism spectrum disorder, and posterior polymorphous corneal dystrophy (PPD). Array-comparative genomic hybridization analysis (Array-CGH) showed a 2.05 Mb de novo interstitial deletion at 10p11.23p11.22. The deleted region overlaps 1 OMIM Morbid Map gene, ZEB1 (the zinc finger E-box binding homeobox transcription factor 1), previously associated with posterior polymorphous corneal dystrophy type 3 (PPCD3). To our best knowledge this is the first reported case with a deletion of the ZEB1 gene in an individual with ACC and PPD, showing that the haploinsufficiency of the ZEB1 is likely the cause of our patient's phenotype.

摘要

我们报告了一名在胎儿超声和磁共振成像检查中被产前诊断为胼胝体发育不全(ACC)的女孩。产后随访发现她存在发育迟缓、面部畸形、自闭症谱系障碍以及后极性多形性角膜营养不良(PPD)。阵列比较基因组杂交分析(Array-CGH)显示在10p11.23p11.22处有一个2.05 Mb的新发间质性缺失。缺失区域与1个OMIM致病图谱基因ZEB1(锌指E盒结合同源框转录因子1)重叠,该基因先前与3型后极性多形性角膜营养不良(PPCD3)相关。据我们所知,这是首例报道的ACC和PPD个体中ZEB1基因缺失的病例,表明ZEB1单倍体不足可能是我们患者表型的原因。

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