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在一个具有常染色体显性高胆固醇血症的巴基斯坦家族中鉴定 LDLR 基因中的一个反复插入突变。

Identification of a recurrent insertion mutation in the LDLR gene in a Pakistani family with autosomal dominant hypercholesterolemia.

机构信息

Department of Biosciences, COMSATS Institute of Information Technology, Park Road, Chak Shahzad, Islamabad, 45600, Pakistan.

出版信息

Mol Biol Rep. 2010 Dec;37(8):3869-75. doi: 10.1007/s11033-010-0043-0. Epub 2010 Mar 10.

DOI:10.1007/s11033-010-0043-0
PMID:20217239
Abstract

Familial Hypercholesterolemia (FH) results in elevated levels of blood lipids including total cholesterol (TC) and low density lipoprotein cholesterol (LDL-C) with normal triglycerides (TG). This disease is one of the major contributors towards an early onset of coronary heart disease (CHD). The aim of the present study was to identify the genes responsible for causing FH in Pakistani population, for this purpose a large consanguineous FH family was selected for genetic analysis. Serum lipid levels, including TC, TG, LDL-C and high density lipoprotein cholesterol (HDL-C), were determined in patients and healthy controls. In order to find the causative mutation in this family, direct sequencing of the low density lipoprotein receptor (LDLR) gene was performed. In addition the part of the Apolipoprotein-B (APOB) gene containing the mutations R3500Q and R3500W was also sequenced. Affected individuals of the family were found to have raised TC and LDL-C levels. Sequencing revealed an insertion mutation (c.2416_2417InsG) in exon 17 of the LDLR gene in all the affected individuals of the family. Common FH causing APOB mutations were not present in this family. Heterozygous individuals had TC levels ranging from ~300-500 mg/dl and the only homozygous individual with typical xanthomas had TC levels exceeding 900 mg/dl. This is the first report of a known LDLR gene mutation causing FH in the Pakistani population. Despite a large heterogeneity of LDLR mutations there are still some common mutations which are responsible for FH throughout the world.

摘要

家族性高胆固醇血症 (FH) 导致血液脂质水平升高,包括总胆固醇 (TC) 和低密度脂蛋白胆固醇 (LDL-C),而甘油三酯 (TG) 正常。这种疾病是导致冠心病 (CHD) 早发的主要原因之一。本研究旨在鉴定导致巴基斯坦人群 FH 的基因,为此选择了一个大型近亲 FH 家族进行遗传分析。在患者和健康对照中测定血清脂质水平,包括 TC、TG、LDL-C 和高密度脂蛋白胆固醇 (HDL-C)。为了在这个家族中找到致病突变,对低密度脂蛋白受体 (LDLR) 基因进行了直接测序。此外,还对载脂蛋白 B (APOB) 基因中包含 R3500Q 和 R3500W 突变的部分进行了测序。该家族的受影响个体 TC 和 LDL-C 水平升高。测序显示 LDLR 基因外显子 17 中的插入突变 (c.2416_2417InsG) 存在于该家族的所有受影响个体中。该家族中不存在常见的引起 FH 的 APOB 突变。杂合子个体的 TC 水平在 ~300-500mg/dl 之间,唯一具有典型黄色瘤的纯合子个体的 TC 水平超过 900mg/dl。这是 LDLR 基因突变导致巴基斯坦人群 FH 的首例报告。尽管 LDLR 突变存在很大的异质性,但仍有一些常见的突变在全球范围内导致 FH。

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Mol Biol Rep. 2011 Jun;38(5):2895-901. doi: 10.1007/s11033-010-9951-2. Epub 2010 Jan 29.
2
Plasma homocysteine and DNA damage profiles in normal and obese subjects in the Pakistani population.巴基斯坦人群中正常和肥胖受试者的血浆同型半胱氨酸水平及DNA损伤情况
Mol Biol Rep. 2010 Jan;37(1):289-95. doi: 10.1007/s11033-009-9686-0. Epub 2009 Aug 8.
3
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Genet Mol Biol. 2018;41(3):570-577. doi: 10.1590/1678-4685-GMB-2016-0287. Epub 2018 Jul 23.
4
Elevated PLA2G7 gene promoter methylation as a gender-specific marker of aging increases the risk of coronary heart disease in females.PLA2G7 基因启动子高甲基化作为一种性别特异性的衰老标志物,增加了女性患冠心病的风险。
PLoS One. 2013;8(3):e59752. doi: 10.1371/journal.pone.0059752. Epub 2013 Mar 28.
5
The genetic spectrum of familial hypercholesterolemia in Pakistan.巴基斯坦家族性高胆固醇血症的遗传谱。
Clin Chim Acta. 2013 Jun 5;421:219-25. doi: 10.1016/j.cca.2013.03.017. Epub 2013 Mar 25.
6
Novel and recurrent LDLR gene mutations in Pakistani hypercholesterolemia patients.巴基斯坦高胆固醇血症患者中新的和反复出现的 LDLR 基因突变。
Mol Biol Rep. 2012 Jul;39(7):7365-72. doi: 10.1007/s11033-012-1568-1. Epub 2012 Feb 7.
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5
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6
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Eur J Hum Genet. 2009 Jan;17(1):85-90. doi: 10.1038/ejhg.2008.138. Epub 2008 Jul 23.
7
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Mol Biol Rep. 2009 May;36(5):1015-21. doi: 10.1007/s11033-008-9275-7. Epub 2008 May 30.
8
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Clin Chim Acta. 2008 Jun;392(1-2):25-9. doi: 10.1016/j.cca.2008.02.019. Epub 2008 Mar 4.
9
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10
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