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病例报告:一名接受体外受精的儿童,患有丙二酰辅酶A脱羧酶缺乏症,其MLYCD基因存在一种新的5'-UTR-外显子1-内含子1缺失及文献综述

Case report: A novel 5'-UTR-exon1-intron1 deletion in MLYCD in an IVF child with malonyl coenzyme A decarboxylase deficiency and literature review.

作者信息

Xu Fang, Wu Yangyang, Huang Jiyi, Zhou Yunguo, Xu Fei, Duan Junkai, Li Hong

机构信息

Cardiology Treatment Center, Jiangxi Provincial Children's Hospital, Nanchang, China.

JXHC Key Laboratory of Children's Cardiovascular Diseases, Jiangxi Provincial Children's Hospital, Nanchang, China.

出版信息

Front Med (Lausanne). 2023 May 3;10:1160879. doi: 10.3389/fmed.2023.1160879. eCollection 2023.

Abstract

The subject of the study is an 11-month old IVF baby girl with the typical clinical manifestation of malonyl coenzyme A decarboxylase deficiency, including developmental delay, limb weakness, cardiomyopathy, and excessive excretion of malonic acid and methylmalonic acid. Whole genome sequencing (WGS) revealed a novel heterozygous nonsense mutation (c.672delG, p.Trp224Ter) in the MLYCD gene of the proband and her father and a novel heterozygous deletion in 5'-UTR-exon1-intron1 of the MLYCD gene of the proband and her mother. The patient's cardiac function and limb weakness improved considerably after 3 months of a low-fat diet supplemented with L-carnitine. Furthermore, mapping of gene mutations and clinical manifestations was done by case collection.

摘要

该研究的对象是一名11个月大的体外受精女婴,具有丙二酰辅酶A脱羧酶缺乏症的典型临床表现,包括发育迟缓、肢体无力、心肌病以及丙二酸和甲基丙二酸排泄过多。全基因组测序(WGS)显示,先证者及其父亲的MLYCD基因存在一个新的杂合无义突变(c.672delG,p.Trp224Ter),先证者及其母亲的MLYCD基因在5'-UTR-外显子1-内含子1存在一个新的杂合缺失。在补充左旋肉碱的低脂饮食3个月后,患者的心脏功能和肢体无力有了显著改善。此外,通过病例收集对基因突变和临床表现进行了定位。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d054/10189016/2fc7b1bf94c7/fmed-10-1160879-g0001.jpg

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