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2
Rare intracranial cholesterol deposition and a homozygous mutation of LDLR in a familial hypercholesterolemia patient.一名家族性高胆固醇血症患者出现罕见的颅内胆固醇沉积及低密度脂蛋白受体纯合突变。
Gene. 2015 Sep 15;569(2):313-7. doi: 10.1016/j.gene.2015.04.071. Epub 2015 Apr 29.
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Novel mutations of low-density lipoprotein receptor gene in China patients with familial hypercholesterolemia.中国家族性高胆固醇血症患者低密度脂蛋白受体基因的新突变
Appl Biochem Biotechnol. 2015 May;176(1):101-9. doi: 10.1007/s12010-015-1554-x. Epub 2015 Apr 7.
4
Genotypic and phenotypic features in homozygous familial hypercholesterolemia caused by proprotein convertase subtilisin/kexin type 9 (PCSK9) gain-of-function mutation.由前蛋白转化酶枯草杆菌蛋白酶/kexin 9型(PCSK9)功能获得性突变引起的纯合子家族性高胆固醇血症的基因型和表型特征。
Atherosclerosis. 2014 Sep;236(1):54-61. doi: 10.1016/j.atherosclerosis.2014.06.005. Epub 2014 Jun 26.
5
Familial hypercholesterolemia in China: prevalence and evidence of underdetection and undertreatment in a community population.中国家族性高胆固醇血症:社区人群中的患病率及漏诊和治疗不足的证据
Int J Cardiol. 2014 Jul 1;174(3):834-6. doi: 10.1016/j.ijcard.2014.04.165. Epub 2014 Apr 21.
6
Functional characterization of ClC-1 mutations from patients affected by recessive myotonia congenita presenting with different clinical phenotypes.对表现出不同临床表型的常染色体隐性先天性肌强直患者的 ClC-1 突变进行功能特征分析。
Exp Neurol. 2013 Oct;248:530-40. doi: 10.1016/j.expneurol.2013.07.018. Epub 2013 Aug 8.
7
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.
8
Identification of LDLR mutations in two Chinese pedigrees with familial hypercholesterolemia.两个中国家族性高胆固醇血症家系中低密度脂蛋白受体(LDLR)突变的鉴定
J Pediatr Endocrinol Metab. 2012;25(7-8):769-73. doi: 10.1515/jpem-2012-0024.
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Homozygous familial hypercholesterolemia: current perspectives on diagnosis and treatment.家族性高胆固醇血症纯合子:诊断与治疗的现状观点。
Atherosclerosis. 2012 Aug;223(2):262-8. doi: 10.1016/j.atherosclerosis.2012.02.019. Epub 2012 Feb 16.
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Combination of two rare mutations causes β-thalassaemia in a Bangladeshi patient.两种罕见突变的组合导致孟加拉国患者患β-地中海贫血。
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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.

DOI:10.3892/etm.2018.6205
PMID:30112042
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC6090432/
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。