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墨西哥非综合征性唇裂患者的IRF6基因多态性

IRF6 polymorphisms in Mexican patients with non-syndromic cleft lip.

作者信息

Ibarra-Arce Aurora, García-Álvarez Martín, Cortés-González Daniel, Ortiz de Zarate-Alarcón Gabriela, Flores-Peña Laura, Sánchez-Camacho Sandra, Arenas-Díaz Silvia, Romero-Valdovinos Mirza, Olivo-Díaz Angélica

机构信息

División de Genética, Hospital General Doctor Manuel Gea González, Mexico DF, Mexico.

Departamento de Biología Molecular e Histocompatibilidad, Hospital General Doctor Manuel Gea González, Mexico DF, Mexico.

出版信息

Meta Gene. 2015 Mar 9;4:8-16. doi: 10.1016/j.mgene.2015.02.002. eCollection 2015 Jun.

Abstract

Cleft lip with or without cleft palate (CL/P) is one of the most common birth defects; it is a multifactorial disease affecting > 1/1,000 live births in Europe, and its etiology is largely unknown, although it is very likely genetic and environmental factors contribute to this malformation. Orofacial development is a complex process involving many genes and signaling pathways. Mutations in the gene for the interferon regulatory factor 6 (IRF6) cause a hereditary dominant malformation syndrome including CL/P, and polymorphisms are associated with non-syndromic CL/P (MIM 119530). Five SNPs at the locus with high heterozygosity in Caucasian populations were chosen for the present research due to their very strong association with CL/P. A case-parent trio study was performed using 292 samples from Mexico. Association with the rs1319435-C/C genotype (P = 0.02) was found in patients (73) as compared to pseudocontrols (219), while the genotype rs1319435-T/C was related with protection (P = 0.041) in the triad design. Significant over-transmission of the G allele for marker rs2235375 (P = 0.049) was found. Only the TACGT haplotype was diminished in the affected child, either in single (P = 0.0208) or double (P = 0.0208) dose. The pairwise analysis showed rs2235543 and rs2235371 were in strong linkage disequilibrium. These results point to a substantial contribution of IRF6 in the etiology of non-syndromic CL/P in a sample of the Mexican population.

摘要

唇裂伴或不伴腭裂(CL/P)是最常见的出生缺陷之一;它是一种多因素疾病,在欧洲每1000例活产中就有超过1例受影响,其病因很大程度上未知,尽管很可能是遗传和环境因素导致了这种畸形。口面部发育是一个涉及许多基因和信号通路的复杂过程。干扰素调节因子6(IRF6)基因的突变会导致一种遗传性显性畸形综合征,包括CL/P,并且多态性与非综合征性CL/P(MIM 119530)相关。由于与CL/P有很强的关联,在白种人群中选择了该位点杂合度高的5个单核苷酸多态性进行本研究。使用来自墨西哥的292个样本进行了病例-父母三联体研究。与假对照(219例)相比,在患者(73例)中发现与rs1319435-C/C基因型相关(P = 0.02),而在三联体设计中,rs1319435-T/C基因型与保护作用相关(P = 0.041)。发现标记rs2235375的G等位基因有显著的过度传递(P = 0.049)。仅TACGT单倍型在患病儿童中无论是单剂量(P = 0.0208)还是双剂量(P = 0.0208)都减少。成对分析表明rs2235543和rs2235371处于强连锁不平衡状态。这些结果表明,在墨西哥人群样本中,IRF6在非综合征性CL/P的病因中起重要作用。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1da4/4354917/760d7f2c1fef/gr1.jpg

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