Kien Nguyen Trung, Nghia Tran Tin, Hoang Nguyen Minh, Phu Tran Nguyen Trong, Nga Pham Thi Ngoc, Mai Ha Thi Thao, Espinoza J Luis
Faculty of Medicine, Can Tho University of Medicine and Pharmacy, Can Tho 900000, Vietnam.
Tra Vinh General Hospital, Tra Vinh 940000, Vietnam.
J Pers Med. 2023 Dec 18;13(12):1725. doi: 10.3390/jpm13121725.
Familial hypercholesterolemia (FH) is an inherited metabolic disorder characterized by high levels of low-density lipoprotein cholesterol (LDL-c) from birth. About 85% of all FH cases are caused by pathogenic variants in the LDLR gene. Individuals with FH have increased cardiovascular risk, including a high risk of premature myocardial infarction (PMI).
We conducted an opportunistic exome screening to identify variants in the LDLR gene among Vietnamese patients with PMI treated at a general hospital in southern Vietnam. A cascade testing for LDLR variants was conducted in their relatives within three generations, and the effects of the LDLR variant on the response to rosuvastatin treatment were also studied using a comparative before-and-after study design on those who were eligible.
A total of 99 participants from the three generations of four PMI patients were recruited, mean age 37.3 ± 18.5 years, 56.6% males. Sanger sequencing revealed two variants in the LDLR gene: variant rs577934998 (c.664T>C), detected in 17 individuals within one family, and variant rs12710260 (c.1060+10G>C), found in 32 individuals (49.5%) in the other three families tested. Individuals harboring the variant c.664T>C had significantly higher baseline LDL-c and total cholesterol levels compared to those with variant c.1060+10G>C (classified as benign) or those without LDLR variants, and among the 47 patients subjected to a 3-month course of rosuvastatin therapy, those with variant c.664T>C had a significantly higher risk of not achieving the LDL-c target after the course of treatment compared to the c.1060+10G>C carriers.
These findings provide evidence supporting the existence of pathogenic LDLR variants in Vietnamese patients with PMI and their relatives and may indicate the need for personalizing lipid-lowering therapies. Further studies are needed to delineate the extent and severity of the problem.
家族性高胆固醇血症(FH)是一种遗传性代谢紊乱疾病,其特征是从出生起低密度脂蛋白胆固醇(LDL-c)水平就很高。所有FH病例中约85%是由LDLR基因的致病变异引起的。FH患者心血管疾病风险增加,包括早发心肌梗死(PMI)的高风险。
我们进行了一次机会性外显子组筛查,以在越南南部一家综合医院接受治疗的PMI越南患者中鉴定LDLR基因的变异。对其三代以内的亲属进行了LDLR变异的级联检测,并采用前后对照研究设计,对符合条件的患者研究了LDLR变异对瑞舒伐他汀治疗反应的影响。
共招募了来自4例PMI患者三代的99名参与者,平均年龄37.3±18.5岁,男性占56.6%。桑格测序在LDLR基因中发现了两个变异:变异rs577934998(c.664T>C),在一个家族的17名个体中检测到;变异rs12710260(c.1060+10G>C),在其他三个检测家族的32名个体(49.5%)中发现。与携带变异c.1060+10G>C(分类为良性)的个体或无LDLR变异的个体相比,携带变异c.664T>C的个体基线LDL-c和总胆固醇水平显著更高,在接受3个月瑞舒伐他汀治疗疗程的47例患者中,与携带c.1060+10G>C变异的个体相比,携带变异c.664T>C的个体在治疗疗程后未达到LDL-c目标的风险显著更高。
这些发现为越南PMI患者及其亲属中存在致病性LDLR变异提供了证据支持,并可能表明需要对降脂治疗进行个性化。需要进一步研究来明确该问题的范围和严重程度。