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先天性肾发育不全的同卵双胞胎的基因组和表观基因组分析。

Genomic and epigenomic analyses of monozygotic twins discordant for congenital renal agenesis.

机构信息

State Key Laboratory of Kidney Disease, Institute of Nephrology, Chinese PLA General Hospital, Beijing, China; Medical College, Nankai University, Tianjin, China.

BGI-Shenzhen, Shenzhen, China.

出版信息

Am J Kidney Dis. 2014 Jul;64(1):119-22. doi: 10.1053/j.ajkd.2014.01.423. Epub 2014 Feb 28.

Abstract

Monozygotic twins have been widely studied to distinguish genetic and environmental factors in the pathogenesis of human diseases. For renal agenesis, the one-sided absence of renal tissue, the relative contributions of genetic and environmental factors to its pathogenesis are still unclear. In this study of a pair of monozygotic twins discordant for congenital renal agenesis, the genomic profile was analyzed from a set of blood samples using high-throughput exome-capture sequencing to detect single-nucleotide polymorphisms (SNPs), copy number variations (CNVs), and insertions and deletions (indels). Also, an epigenomic analysis used reduced-representation bisulfite sequencing to detect differentially methylated regions (DMRs). No discordant SNPs, CNVs, or indels were confirmed, but 514 DMRs were detected. KEGG analysis indicated the DMRs localized to 10 signaling pathways and 25 genes, including the mitogen-activated protein kinase pathway and 6 genes (FGF18, FGF12, PDGFRA, MAPK11, AMH, CTBP1) involved in organ development. Although methylation results from our adult patient and her sister may not represent the pattern that was present during kidney development, we could at least confirm a lack of obvious differences at the genome level, which suggests that nongenetic factors may be involved in the pathogenesis of renal agenesis.

摘要

同卵双胞胎被广泛研究,以区分人类疾病发病机制中的遗传和环境因素。对于肾发育不全,单侧肾组织缺失,遗传和环境因素对其发病机制的相对贡献仍不清楚。在这项对一对先天性肾发育不全的同卵双胞胎的研究中,使用高通量外显子组捕获测序从一组血液样本中分析基因组图谱,以检测单核苷酸多态性(SNP)、拷贝数变异(CNV)和插入缺失(indel)。此外,还进行了表观基因组分析,使用简化代表性亚硫酸氢盐测序来检测差异甲基化区域(DMR)。未确认存在不一致的 SNP、CNV 或 indel,但检测到 514 个 DMR。KEGG 分析表明,DMR 定位于 10 个信号通路和 25 个基因,包括丝裂原活化蛋白激酶途径和 6 个参与器官发育的基因(FGF18、FGF12、PDGFRA、MAPK11、AMH、CTBP1)。尽管我们的成年患者及其妹妹的甲基化结果可能不能代表肾脏发育过程中的模式,但我们至少可以确认在基因组水平上没有明显差异,这表明非遗传因素可能参与了肾发育不全的发病机制。

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