Department of Medical Microbiology and Immunology, MIND Institute, Genome Center, University of California, Davis, CA 95616, USA.
Cereb Cortex. 2020 Mar 21;30(2):640-655. doi: 10.1093/cercor/bhz115.
Neurodevelopmental disorders (NDDs) affect 7-14% of all children in developed countries and are one of the leading causes of lifelong disability. Epigenetic modifications are poised at the interface between genes and environment and are predicted to reveal insight into NDD etiology. Whole-genome bisulfite sequencing was used to examine DNA cytosine methylation in 49 human cortex samples from 3 different NDDs (autism spectrum disorder, Rett syndrome, and Dup15q syndrome) and matched controls. Integration of methylation changes across NDDs with relevant genomic and genetic datasets revealed differentially methylated regions (DMRs) unique to each type of NDD but with shared regulatory functions in neurons and microglia. NDD DMRs were enriched within promoter regions and for transcription factor binding sites with identified methylation sensitivity. DMRs from all 3 disorders were enriched for ontologies related to nervous system development and genes with disrupted expression in brain from neurodevelopmental or neuropsychiatric disorders. Genes associated with NDD DMRs showed expression patterns indicating an important role for altered microglial function during brain development. These findings demonstrate an NDD epigenomic signature in human cortex that will aid in defining therapeutic targets and early biomarkers at the interface of genetic and environmental NDD risk factors.
神经发育障碍(NDD)影响发达国家所有儿童的 7-14%,是导致终身残疾的主要原因之一。表观遗传修饰处于基因与环境之间的界面,预计能深入了解 NDD 的病因。全基因组亚硫酸氢盐测序用于研究来自 3 种不同 NDD(自闭症谱系障碍,雷特综合征和 Dup15q 综合征)和匹配对照的 49 个人类大脑皮质样本中的 DNA 胞嘧啶甲基化。将 NDD 中的甲基化变化与相关的基因组和遗传数据集进行整合,揭示了每种 NDD 特有的差异甲基化区域(DMR),但在神经元和小胶质细胞中具有共同的调节功能。NDD DMR 富集在启动子区域内,并且在转录因子结合位点上具有确定的甲基化敏感性。所有 3 种疾病的 DMR 都富含与神经系统发育相关的本体论和与神经发育或神经精神疾病有关的大脑中表达中断的基因。与 NDD DMR 相关的基因表现出表达模式,表明在大脑发育过程中改变小胶质细胞功能具有重要作用。这些发现证明了人类大脑皮层中的 NDD 表观基因组特征,这将有助于确定治疗靶点和遗传与环境 NDD 风险因素界面的早期生物标志物。