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通过全外显子组测序鉴定一名中国科恩综合征患者中的新型VPS13B突变

Identification of a Novel VPS13B Mutation in a Chinese Patient with Cohen Syndrome by Whole-Exome Sequencing.

作者信息

Hu Xiaoyun, Huang Tao, Liu Yun, Zhang Lina, Zhu Li, Peng Xiaohong, Zhang Sufang

机构信息

Department of Pediatrics, The First Affiliated Hospital of Nanchang University, Nanchang, People's Republic of China.

出版信息

Pharmgenomics Pers Med. 2021 Dec 4;14:1583-1589. doi: 10.2147/PGPM.S327252. eCollection 2021.

Abstract

OBJECTIVE

The present study aims to investigate the clinical features and diagnostic characteristics of children with Cohen syndrome caused by the vacuolar protein sorting 13 homolog B (VPS13B) gene mutation and to review the relevant literature to provide a reference for genetic counseling and the diagnosis of Cohen syndrome.

METHODS

The clinical data and molecular genetic test results of a child with Cohen syndrome were retrospectively analyzed and a review of the relevant literature was conducted.

RESULTS

A two-year-and-four-month-old boy was referred to the hospital for recurrent fever and shortness of breath. On physical examination, the boy was found to have growth retardation, thick bushy hair, microcephaly, hypertelorism, down-slanting palpebral fissures, and hypotonia. Genetic testing was performed, and the results suggested the presence of exon 20-32 heterozygous deletion and c.8275 delC (p.R2759 fs*18) heterozygous variant on the VPS13B gene from phenotypically normal parents. These two mutation loci have not been reported in the literature, and they were predicted by relevant software to be pathogenic variants.

CONCLUSION

We identified two novel variants in the VPS13B gene (exon 20-32 heterozygous deletion and c.8275 delC heterozygous variant) in a boy with Cohen syndrome, thus extending the spectrum of VPS13B gene variants in patients with Cohen syndrome.

摘要

目的

本研究旨在探讨由空泡蛋白分选13同源物B(VPS13B)基因突变引起的科恩综合征患儿的临床特征和诊断特点,并复习相关文献,为遗传咨询和科恩综合征的诊断提供参考。

方法

回顾性分析1例科恩综合征患儿的临床资料及分子遗传学检测结果,并对相关文献进行复习。

结果

一名2岁4个月大的男孩因反复发热和呼吸急促被转诊至我院。体格检查发现该男孩存在生长发育迟缓、头发浓密且粗、小头畸形、眼距增宽、睑裂向下倾斜及肌张力低下。进行了基因检测,结果显示来自表型正常父母的VPS13B基因存在外显子20 - 32杂合缺失和c.8275 delC(p.R2759 fs*18)杂合变异。这两个突变位点在文献中尚未见报道,相关软件预测它们为致病变异。

结论

我们在1例科恩综合征男孩中鉴定出VPS13B基因的两个新变异(外显子20 - 32杂合缺失和c.8275 delC杂合变异),从而扩展了科恩综合征患者VPS13B基因变异谱。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bd7e/8657012/b10b72a91642/PGPM-14-1583-g0001.jpg

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