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全外显子组测序鉴定 1 例中国新生儿肝衰竭的新型复合杂合变异及文献复习

Whole-exome sequencing identified novel compound heterozygous variants in a Chinese neonate with liver failure and review of literature.

机构信息

Genetic and Metabolic Central Laboratory, Guangxi Birth Defects Research and Prevention Institute, Maternal and Child Health Hospital of Guangxi Zhuang Autonomous Region, Nanning, Guangxi, China.

Department of Genetics, Harvard Medical School, Boston, MA, USA.

出版信息

Mol Genet Genomic Med. 2020 Dec;8(12):e1515. doi: 10.1002/mgg3.1515. Epub 2020 Nov 18.

Abstract

BACKGROUND

Liver failure caused by TRMU is a rare hereditary disorder and clinically manifests into metabolic acidosis, hyperlactatemia, and hypoglycemia. Limited spectrum of TRMU pathogenic variants has been reported.

METHODS

Whole-exome sequencing was employed for the diagnosis of a 5-day-old female who suffered from severe neonatal hyperlactatemia and hypoglycemia since birth. Sanger sequencing was performed to confirm the origin of the variants subsequently. Variants classification was followed to ACMG guideline.

RESULTS

A compound heterozygosity of a frameshiftc.34_35dupTC (p.Gly13fs) and a missense c.244T>G (p.Phe82Val) in TRMU was detected, both variants are novel and pathogenic. Analysis of clinical and genetic information including patients reported previously indicated that there is no significant correlation between the genotype and the phenotype of TRMU-caused liver failure.

CONCLUSION

To the best of our knowledge, this is the first case report of TRMU-caused liver failure in China. Whole-exome sequencing is effective for conclusive diagnosis of this disorder and beneficial for its clinical management.

摘要

背景

TRMU 导致的肝衰竭是一种罕见的遗传性疾病,临床上表现为代谢性酸中毒、高乳酸血症和低血糖症。已有报道称 TRMU 致病性变异体的范围有限。

方法

对一名出生 5 天的女性进行全外显子组测序,该女性自出生以来即患有严重的新生儿高乳酸血症和低血糖症。随后进行 Sanger 测序以确认变异体的来源。根据 ACMG 指南对变异体进行分类。

结果

检测到 TRMU 中存在复合杂合的移码 c.34_35dupTC(p.Gly13fs)和错义 c.244T>G(p.Phe82Val),这两种变异均为新的致病性变异。对包括先前报道的患者在内的临床和遗传信息进行分析表明,TRMU 引起的肝衰竭的基因型与表型之间没有明显相关性。

结论

据我们所知,这是中国首例 TRMU 引起的肝衰竭病例报告。全外显子组测序是该疾病明确诊断的有效方法,有利于其临床管理。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b78c/7767550/1087b76ee766/MGG3-8-e1515-g001.jpg

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