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在一个胎儿中发现 NPR2 基因的新 c.2455C>T 突变,该胎儿长骨缩短并伴有室间隔缺损,其母亲在 16q22.1 处具有脆弱部位,父亲在越南具有 4 号染色体罕见异染色质变异。

De novo c.2455C>T mutation of NPR2 gene in a fetus with shortened long bones and a ventricular septal defect conceived by a mother with a fragile site at 16q22.1 and a father with a rare heterochromatic variant of chromosome 4 from Vietnam.

机构信息

Department of Medical Genetics, Hue University of Medicine and Pharmacy, Hue University, Hue, Vietnam.

Center of Prenatal and Neonatal Screening-Diagnosis, Hue University of Medicine and Pharmacy Hospital, Hue, Vietnam.

出版信息

Mol Genet Genomic Med. 2021 Apr;9(4):e1637. doi: 10.1002/mgg3.1637. Epub 2021 Mar 13.

Abstract

BACKGROUND

A heterozygous natriuretic peptide receptor 2 (NPR2) gene c.2455C>T mutation was identified as a cause of familial idiopathic short stature (ISS). Only two cases with this mutation were reported previously, and the probands with ISS had no organ system defects.

METHODS

Next-generation sequencing (NGS) was performed on an amniotic fluid DNA sample of a fetus with shortened long bones and a small ventricular septal defect detected by an obstetric ultrasound examination. The pathogenic variant of the fetus was confirmed by Sanger sequencing. Sanger sequencing, G-banded, and C-banded karyotyping of the fetus's parents were subsequently performed.

RESULTS

A de novo NPR2 gene c.2455C>T, p.(Arg819Cys) mutation was identified in the fetus. No microdeletion or microduplication was identified in the fetus by copy number variation sequencing with a maximum resolution of 400 kb. The two previous miscarriages experienced by the fetus's parents were interpreted as a result of chromosomal aberrations, including a maternal fragile site at 16q22.1 and a rare paternal variant involving in a large G-band-positive and C-band-positive block of paracentric heterochromatin of chromosome 4p.

CONCLUSION

This report provides clinical signs of a de novo heterozygous NPR2 gene c.2455C>T mutation in the fetus and shows paternal chromosomal aberrations causing repeated pregnancy loss.

摘要

背景

杂合性利钠肽受体 2(NPR2)基因 c.2455C>T 突变被认为是家族性特发性身材矮小(ISS)的病因。此前仅报道过两例携带该突变的病例,ISS 先证者无器官系统缺陷。

方法

对经产科超声检查发现长骨缩短和小室间隔缺损的胎儿羊水 DNA 样本进行下一代测序(NGS)。通过 Sanger 测序确认胎儿的致病性变异。随后对胎儿父母进行 Sanger 测序、G 带和 C 带核型分析。

结果

在胎儿中发现了 NPR2 基因 c.2455C>T,p.(Arg819Cys)的新生突变。通过最大分辨率为 400kb 的拷贝数变异测序,未在胎儿中发现微缺失或微重复。胎儿父母的两次先前流产被解释为染色体异常的结果,包括母体 16q22.1 脆性位点和罕见的父亲变异,涉及染色体 4p 着丝粒旁异染色质的大片段 G 带阳性和 C 带阳性。

结论

本报告提供了胎儿 NPR2 基因 c.2455C>T 新生杂合突变的临床特征,并显示了导致反复妊娠丢失的父本染色体异常。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9c43/8123736/e925d8ed3330/MGG3-9-e1637-g001.jpg

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