Li Jinchen, Cai Tao, Jiang Yi, Chen Huiqian, He Xin, Chen Chao, Li Xianfeng, Shao Qianzhi, Ran Xia, Li Zhongshan, Xia Kun, Liu Chunyu, Sun Zhong Sheng, Wu Jinyu
Beijing Institutes of Life Science, Chinese Academy of Sciences, Beijing, China.
Institute of Genomic Medicine, Wenzhou Medical University, Wenzhou, China.
Mol Psychiatry. 2016 Feb;21(2):290-7. doi: 10.1038/mp.2015.40. Epub 2015 Apr 7.
Currently, many studies on neuropsychiatric disorders have utilized massive trio-based whole-exome sequencing (WES) and whole-genome sequencing (WGS) to identify numerous de novo mutations (DNMs). Here, we retrieved 17,104 DNMs from 3555 trios across four neuropsychiatric disorders: autism spectrum disorder, epileptic encephalopathy, intellectual disability and schizophrenia, in addition to unaffected siblings (control), from 36 studies by WES/WGS. After eliminating non-exonic variants, we focused on 3334 exonic DNMs for evaluation of their association with these diseases. Our results revealed a higher prevalence of DNMs in the probands of all four disorders compared with the one in the controls (P<1.3 × 10(-7)). The elevated DNM frequency is dominated by loss-of-function/deleterious single-nucleotide variants and frameshift indels (that is, extreme mutations, P<4.5 × 10(-5)). With extensive annotation of these 'extreme' mutations, we prioritized 764 candidate genes in these four disorders. A combined analysis of Gene Ontology, microRNA targets and transcription factor targets revealed shared biological process and non-coding regulatory elements of candidate genes in the pathology of neuropsychiatric disorders. In addition, weighted gene co-expression network analysis of human laminar-specific neocortical expression data showed that candidate genes are convergent on eight shared modules with specific layer enrichment and biological process features. Furthermore, we identified that 53 candidate genes are associated with more than one disorder (P<0.000001), suggesting a possibly shared genetic etiology underlying these disorders. Particularly, DNMs of the SCN2A gene are frequently occurred across all four disorders. Finally, we constructed a freely available NPdenovo database, which provides a comprehensive catalog of the DNMs identified in neuropsychiatric disorders.
目前,许多关于神经精神疾病的研究利用了大量基于三联体的全外显子组测序(WES)和全基因组测序(WGS)来识别众多新生突变(DNM)。在此,我们从36项采用WES/WGS的研究中,检索了来自3555个三联体的17104个DNM,这些三联体涉及四种神经精神疾病:自闭症谱系障碍、癫痫性脑病、智力障碍和精神分裂症,以及未受影响的同胞(对照)。在剔除非外显子变异后,我们聚焦于3334个外显子DNM来评估它们与这些疾病的关联。我们的结果显示,与对照组相比,所有四种疾病的先证者中DNM的患病率更高(P<1.3×10⁻⁷)。DNM频率的升高主要由功能丧失/有害单核苷酸变异和移码插入缺失(即极端突变,P<4.5×10⁻⁵)主导。通过对这些“极端”突变进行广泛注释,我们在这四种疾病中确定了764个候选基因。基因本体论、微小RNA靶标和转录因子靶标的综合分析揭示了候选基因在神经精神疾病病理学中的共同生物学过程和非编码调控元件。此外,对人类层特异性新皮质表达数据进行加权基因共表达网络分析表明,候选基因汇聚在八个具有特定层富集和生物学过程特征的共享模块中。此外,我们确定53个候选基因与不止一种疾病相关(P<0.000001),这表明这些疾病可能存在共同的遗传病因。特别是,SCN2A基因的DNM在所有四种疾病中都频繁出现。最后我们构建了一个免费可用的NPdenovo数据库,该数据库提供了在神经精神疾病中鉴定出的DNM的综合目录。