Department of Neurology, School of Clinical Medicine, the Affiliated Hospital of Putian UniversityFujian Medical UniversityBrain Science Institute of Putian University, 999 Dongzhen East Road, Licheng District, Putian, 351100, China.
Neurogenetics. 2024 Apr;25(2):131-139. doi: 10.1007/s10048-024-00748-w. Epub 2024 Mar 9.
Twin and family studies have established the genetic contribution to idiopathic generalized epilepsy (IGE). The genetic architecture of IGE is generally complex and heterogeneous, and the majority of the genetic burden in IGE remains unsolved. We hypothesize that gene-gene interactions contribute to the complex inheritance of IGE. CNTN2 (OMIM* 615,400) variants have been identified in cases with familial adult myoclonic epilepsy and other epilepsies. To explore the gene-gene interaction network in IGE, we took the CNTN2 gene as an example and investigated its co-occurrent genetic variants in IGE cases. We performed whole-exome sequencing in 114 unrelated IGE cases and 296 healthy controls. Variants were qualified with sequencing quality, minor allele frequency, in silico prediction, genetic phenotype, and recurrent case numbers. The STRING_TOP25 gene interaction network analysis was introduced with the bait gene CNTN2 (denoted as A). The gene-gene interaction pair mode was presumed to be A + c, A + d, A + e, with a leading gene A, or A + B + f, A + B + g, A + B + h, with a double-gene A + B, or other combinations. We compared the number of gene interaction pairs between the case and control groups. We identified three pairs in the case group, CNTN2 + PTPN18, CNTN2 + CNTN1 + ANK2 + ANK3 + SNTG2, and CNTN2 + PTPRZ1, while we did not discover any pairs in the control group. The number of gene interaction pairs in the case group was much more than in the control group (p = 0.021). Taking together the genetic bioinformatics, reported epilepsy cases, and statistical evidence in the study, we supposed CNTN2 as a candidate pathogenic gene for IGE. The gene interaction network analysis might help screen candidate genes for IGE or other complex genetic disorders.
双生子和家系研究已经确定了特发性全面性癫痫(IGE)的遗传贡献。IGE 的遗传结构通常较为复杂且异质,IGE 的大部分遗传负担仍未得到解决。我们假设基因-基因相互作用导致 IGE 的复杂遗传。已经在家族性成年肌阵挛性癫痫和其他癫痫病例中鉴定出 CNTN2(OMIM*615400)变体。为了探索 IGE 中的基因-基因相互作用网络,我们以 CNTN2 基因为例,研究了其在 IGE 病例中的并发遗传变体。我们对 114 例无关的 IGE 病例和 296 例健康对照进行了全外显子组测序。变体经过测序质量、次要等位基因频率、计算机预测、遗传表型和反复出现的病例数量等方面的筛选。引入 STRING_TOP25 基因相互作用网络分析,以 CNTN2 基因为诱饵基因(表示为 A)。基因-基因相互作用对模式被假定为 A+c、A+d、A+e,以主导基因 A 或 A+B+f、A+B+g、A+B+h,以双基因 A+B 或其他组合。我们比较了病例组和对照组中基因相互作用对的数量。在病例组中鉴定出三个对,即 CNTN2+PTPN18、CNTN2+CNTN1+ANK2+ANK3+SNTG2 和 CNTN2+PTPRZ1,而在对照组中没有发现任何对。病例组中基因相互作用对的数量明显多于对照组(p=0.021)。综合遗传生物信息学、报道的癫痫病例和研究中的统计证据,我们推测 CNTN2 是 IGE 的候选致病基因。基因相互作用网络分析可能有助于筛选 IGE 或其他复杂遗传疾病的候选基因。