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通过产前全外显子组测序鉴定了 3 例先天性脑积水胎儿中的 FKTN、POMGNT1 和 LAMB1 基因的复合变异。

Compound variants of FKTN, POMGNT1, and LAMB1 gene identified by prenatal whole-exome sequencing in three fetuses with congenital hydrocephalus.

机构信息

Guangxi Center for Birth Defects Research and Prevention, Maternal and Child Health Hospital of Guangxi Zhuang Autonomous Region, Nanning, Guangxi, P.R. China.

Department of Clinical Genetics, Maternal and Child Health Hospital of Guangxi Zhuang Autonomous Region, Nanning, Guangxi, P.R. China.

出版信息

J Obstet Gynaecol Res. 2022 Oct;48(10):2624-2629. doi: 10.1111/jog.15358. Epub 2022 Jul 17.

Abstract

Congenital hydrocephalus (CH) is a severe birth defect, and genetics components is an important etiology. Whole-exome sequencing (WES) has been proven to be a feasible approach for prenatal diagnosis of CH. In this study, we carried out WES on three fetuses with cerebral ventriculomegaly. After bioinformation analysis and data filtering, three compound variants, c.919C>T(p.Arg307Ter)/c.1100del(p.Phe369fs) in FKTN, c.1449_1450insACAACG/c.1490G>C(p.Arg497Pro) in POMGNT1, and c.2690+1G>A/c.1447C>T(p.Arg483Cys) in LAMB1 were detected in the three fetuses. All the six variants were classified as likely pathogenic or pathogenic in accordance with the American College of Medical Genetics and Genomics-Association for Molecular Pathology guidelines. This study provides support for the potential of WES for the accurate prenatal diagnosis of fetal hydrocephalus and further demonstrated the genetic heterogeneity in patients with CH. The novel variants (c.1449_1450insACAACG and c.1490G>C in POMGNT1, c.2690+1G>A in LAMB1) expanded the gene mutational spectrum of CH and contributes to genetics counseling and pregnancy management.

摘要

先天性脑积水(CH)是一种严重的出生缺陷,遗传因素是其重要病因之一。全外显子组测序(WES)已被证明是产前诊断 CH 的一种可行方法。本研究对 3 例脑室扩张的胎儿进行了 WES 分析。经过生物信息分析和数据过滤,在这 3 例胎儿中检测到了 FKTN 基因中的 c.919C>T(p.Arg307Ter)/c.1100del(p.Phe369fs)、POMGNT1 基因中的 c.1449_1450insACAACG/c.1490G>C(p.Arg497Pro)和 LAMB1 基因中的 c.2690+1G>A/c.1447C>T(p.Arg483Cys)这 3 个复合变异。根据美国医学遗传学与基因组学学会-分子病理学指南,这 6 个变异均被归类为可能致病或致病性变异。本研究为 WES 准确产前诊断胎儿脑积水提供了依据,进一步证实了 CH 患者的遗传异质性。这些新的变异(POMGNT1 基因中的 c.1449_1450insACAACG 和 c.1490G>C,LAMB1 基因中的 c.2690+1G>A)扩展了 CH 的基因突变谱,有助于遗传咨询和妊娠管理。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4b59/9796612/80cca0218f86/JOG-48-2624-g001.jpg

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