Arai Yoshikazu, Umeyama Kazuhiro, Takeuchi Kenta, Okazaki Natsumi, Hichiwa Naomi, Yashima Sayaka, Nakano Kazuaki, Nagashima Hiroshi, Ohgane Jun
Laboratory of Genomic Function Engineering, Department of Life Sciences, School of Agriculture, Meiji University, Kanagawa 214-8571, Japan.
Laboratory of Veterinary Biochemistry and Molecular Biology, Faculty of Agriculture, University of Miyazaki, Miyazaki 889-2192, Japan.
J Reprod Dev. 2017 Apr 21;63(2):157-165. doi: 10.1262/jrd.2016-158. Epub 2017 Jan 22.
DNA methylation in transcriptional regulatory regions is crucial for gene expression. The DNA methylation status of the edges of CpG islands, called CpG island shore, is involved in tissue/cell-type-specific gene expression. Haploinsufficiency diseases are caused by inheritance of one mutated null allele and are classified as autosomal dominant. However, in the same pedigree, phenotypic variances are observed despite the inheritance of the identical mutated null allele, including Fibrillin1 (FBN1), which is responsible for development of the haploinsufficient Marfan disease. In this study, we examined the relationship between gene expression and DNA methylation patterns of the FBN1 CpG island shore focusing on transcriptionally active hypomethylated alleles (Hypo-alleles). No difference in the DNA methylation level of FBN1 CpG island shore was observed in porcine fetal fibroblast (PFF) and the liver, whereas FBN1 expression was higher in PFF than in the liver. However, Hypo-allele ratio of the FBN1 CpG island shore in PFF was higher than that in the liver, indicating that Hypo-allele ratio of the FBN1 CpG island shore likely correlated with FBN1 expression level. In addition, oocyte-derived DNA hypermethylation in preimplantation embryos was erased until the blastocyst stage, and re-methylation of the FBN1 CpG island shore was observed with prolonged in vitro culture of blastocysts. These results suggest that the establishment of the DNA methylation pattern within the FBN1 CpG island shore occurs after the blastocyst stage, likely during organogenesis. In conclusion, Hypo-allele ratios of the FBN1 CpG island shore correlated with FBN1 expression levels in porcine tissues.
转录调控区域的DNA甲基化对基因表达至关重要。CpG岛边缘的DNA甲基化状态,即所谓的CpG岛岸,参与组织/细胞类型特异性基因表达。单倍剂量不足疾病是由一个突变的无效等位基因的遗传引起的,被归类为常染色体显性遗传。然而,在同一个家系中,尽管继承了相同的突变无效等位基因,包括导致单倍剂量不足的马凡综合征的原纤维蛋白1(FBN1),仍观察到表型差异。在本研究中,我们聚焦于转录活性的低甲基化等位基因(低甲基化等位基因),研究了FBN1 CpG岛岸的基因表达与DNA甲基化模式之间的关系。在猪胎儿成纤维细胞(PFF)和肝脏中,未观察到FBN1 CpG岛岸的DNA甲基化水平存在差异,而FBN1在PFF中的表达高于肝脏。然而,PFF中FBN1 CpG岛岸的低甲基化等位基因比例高于肝脏,这表明FBN1 CpG岛岸的低甲基化等位基因比例可能与FBN1表达水平相关。此外,植入前胚胎中卵母细胞来源的DNA高甲基化在囊胚阶段之前被消除,并且随着囊胚体外培养时间的延长,观察到FBN1 CpG岛岸的重新甲基化。这些结果表明,FBN1 CpG岛岸内的DNA甲基化模式在囊胚阶段之后建立,可能发生在器官发生过程中。总之,FBN1 CpG岛岸的低甲基化等位基因比例与猪组织中的FBN1表达水平相关。