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低密度脂蛋白受体基因中的新型复合杂合突变在中国一个高胆固醇血症家族中导致严重表型。

Novel compound heterozygous mutations in low density lipoprotein receptor gene causes a severe phenotype in a Chinese hypercholesterolemia family.

作者信息

Cheng Xinyao, Huang Yifang, Qiu Xueping, Cheng Xiaohuan, Jin Yalei, Hu Yafei, Yang Bing, Zhao Jingbo, Lei Yuhua, Zheng Fang

机构信息

Cardiovascular Division, Zhongnan Hospital, Wuhan University, Wuhan, Hubei 430071, P.R. China.

Center for Gene Diagnosis, Zhongnan Hospital, Wuhan University, Wuhan, Hubei 430071, P.R. China.

出版信息

Exp Ther Med. 2018 Aug;16(2):901-907. doi: 10.3892/etm.2018.6205. Epub 2018 May 23.

Abstract

Mutations in the low density lipoprotein receptor (LDLR) gene serve a causative role in the pathophysiology of familial hypercholesterolemia (FH), a common autosomal inherited disorder characterized by abnormal lipid metabolism. The aim of the present study was to investigate genetic defects in a Chinese family with FH. Clinical features and family histories were collected, as were the results of various laboratory tests, including determinations of serum lipid concentrations, ultrasonography and angiography results. Potential mutations in LDLR were screened using direct polymerase chain reaction (PCR) sequencing. Multiple sequence alignments, structure and hydrophobicity predictions were performed in silico. Novel compound heterozygote mutations in LDLR of the proband were identified, with a Trp577Term-bearing maternal allele and a Pro685Leu-bearing paternal allele. The proband, a 27-year-old male, had severe and diffuse coronary stenosis and non-ST segment elevation myocardial infarction, as well as multiple skin xanthomas and high serum lipid levels. The allele-dosage-dependent clinical features, including hypercholesterolemia and peripheral arterial atherosclerosis, were observed in the proband and the other heterozygous patients. Therefore, the coexistence of Pro685Leu and Trp577Term mutations in LDLR is a novel compound heterozygosis in Chinese patients and may lead to a severe FH phenotype. The explanation for the existence of compound heterozygous mutations instead of homozygous mutations in this particular family requires further study.

摘要

低密度脂蛋白受体(LDLR)基因突变在家族性高胆固醇血症(FH)的病理生理学中起致病作用,FH是一种常见的常染色体显性遗传病,其特征为脂质代谢异常。本研究的目的是调查一个中国FH家族中的基因缺陷。收集了临床特征和家族病史,以及各种实验室检查结果,包括血脂浓度测定、超声检查和血管造影结果。使用直接聚合酶链反应(PCR)测序筛选LDLR中的潜在突变。在计算机上进行了多序列比对、结构和疏水性预测。在先证者的LDLR中鉴定出新型复合杂合突变,其携带Trp577Term的母本等位基因和携带Pro685Leu的父本等位基因。该先证者为一名27岁男性,患有严重弥漫性冠状动脉狭窄和非ST段抬高型心肌梗死,以及多处皮肤黄色瘤和高血脂水平。在先证者和其他杂合患者中观察到等位基因剂量依赖性临床特征,包括高胆固醇血症和外周动脉粥样硬化。因此,LDLR中Pro685Leu和Trp577Term突变的共存是中国患者中的一种新型复合杂合状态,可能导致严重的FH表型。对于这个特定家族中存在复合杂合突变而非纯合突变的原因需要进一步研究。

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Diagnosis and treatment of familial hypercholesterolaemia.家族性高胆固醇血症的诊断和治疗。
Eur Heart J. 2013 Apr;34(13):962-71. doi: 10.1093/eurheartj/eht015. Epub 2013 Feb 14.

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