Kuehl Karen, Loffredo Christopher, Lammer Edward J, Iovannisci David M, Shaw Gary M
Children's National Heart Institute, Washington, DC 20010, USA.
Birth Defects Res A Clin Mol Teratol. 2010 Feb;88(2):101-10. doi: 10.1002/bdra.20630.
Clark (1996) proposed that abnormal blood flow is related to some congenital cardiovascular malformations (CCVMs), particularly CCVM with obstruction to blood flow. Our hypothesis is that CCVMs may relate to genes that affect blood coagulation or flow. We studied whether polymorphisms of such genes are related to CCVMs; previous association of these SNPs to conotruncal CCVMs is described.
We assessed risk of pulmonary stenosis (PS, N = 120), atrial septal defect (ASD, N = 108), aortic stenosis (AS, N = 36), and coarctation of the aorta (CoAo, N = 64), associated with 33 candidate genes, selected for their relationship to blood flow affected by homocysteine metabolism, coagulation, cell-cell interaction, inflammation, or blood pressure regulation.
Effects were specific to cardiac phenotype and race. CoAo was associated with MTHFR (-667) C>T (odds ratio [OR] for TT 3.5, 95% confidence limits [CI] 1.4-8.6). AS was associated with a polymorphism of SERPINE1, G5>G4, OR = 5.6 for the homozygote with 95% CI 1.4-22.9. Unique polymorphisms were associated with increased risk of ASD and PS: NPPA 664G>A with ASD (OR of 2.4, 95%CI 1.3-4.4) and NOS3 (-690) C>T with PS (OR 6.1; 95% CI 1.6-22.6 in the African American population only). For ASD, the NPPA (-664) G>A SNP there was increased risk from the variant genotype only in maternal smokers (OR 2.6; 95% CI 1.0-7.2).
Genes affecting vascular function and coagulation appear to be promising candidates for the etiology of cardiac malformations and warrant further study.
克拉克(1996年)提出,异常血流与一些先天性心血管畸形(CCVMs)有关,特别是伴有血流梗阻的CCVMs。我们的假设是,CCVMs可能与影响血液凝固或血流的基因有关。我们研究了此类基因的多态性是否与CCVMs有关;此前已描述了这些单核苷酸多态性(SNPs)与圆锥动脉干CCVMs的关联。
我们评估了与33个候选基因相关的肺动脉狭窄(PS,N = 120)、房间隔缺损(ASD,N = 108)、主动脉狭窄(AS,N = 36)和主动脉缩窄(CoAo,N = 64)的风险,这些候选基因因其与同型半胱氨酸代谢、凝血、细胞间相互作用、炎症或血压调节所影响的血流的关系而被选中。
效应因心脏表型和种族而异。CoAo与亚甲基四氢叶酸还原酶(MTHFR)(-667)C>T相关(TT的优势比[OR]为3.5,95%置信区间[CI]为1.4 - 8.6)。AS与丝氨酸蛋白酶抑制剂1(SERPINE1)的一种多态性G5>G4相关,纯合子的OR = 5.6,95%CI为1.4 - 22.9。独特的多态性与ASD和PS风险增加相关:NPPA 664G>A与ASD相关(OR为2.4,95%CI为1.3 - 4.4),NOS3(-690)C>T与PS相关(仅在非裔美国人中OR为6.1;95%CI为1.6 - 22.6)。对于ASD,仅在母亲吸烟的情况下,NPPA(-664)G>A SNP的变异基因型风险增加(OR为2.6;95%CI为1.0 - 7.2)。
影响血管功能和凝血的基因似乎是心脏畸形病因学中有前景的候选基因,值得进一步研究。