Li Rudong, Reiter Jill L, Chen Andy B, Chen Steven X, Foroud Tatiana, Edenberg Howard J, Lai Dongbing, Liu Yunlong
Center for Computational Biology and Bioinformatics, Indiana University School of Medicine, Indianapolis, IN, 46202, USA.
Department of Medical and Molecular Genetics, Indiana University School of Medicine, Indianapolis, IN, 46202, USA.
Mol Psychiatry. 2023 Jul;28(7):2922-2933. doi: 10.1038/s41380-023-02111-1. Epub 2023 May 23.
Alcohol use disorder (AUD) is a complex genetic disorder characterized by problems arising from excessive alcohol consumption. Identifying functional genetic variations that contribute to risk for AUD is a major goal. Alternative splicing of RNA mediates the flow of genetic information from DNA to gene expression and expands proteome diversity. We asked whether alternative splicing could be a risk factor for AUD. Herein, we used a Mendelian randomization (MR)-based approach to identify skipped exons (the predominant splicing event in brain) that contribute to AUD risk. Genotypes and RNA-seq data from the CommonMind Consortium were used as the training dataset to develop predictive models linking individual genotypes to exon skipping in the prefrontal cortex. We applied these models to data from the Collaborative Studies on Genetics of Alcoholism to examine the association between the imputed cis-regulated splicing outcome and the AUD-related traits. We identified 27 exon skipping events that were predicted to affect AUD risk; six of these were replicated in the Australian Twin-family Study of Alcohol Use Disorder. Their host genes are DRC1, ELOVL7, LINC00665, NSUN4, SRRM2 and TBC1D5. The genes downstream of these splicing events are enriched in neuroimmune pathways. The MR-inferred impacts of the ELOVL7 skipped exon on AUD risk was further supported in four additional large-scale genome-wide association studies. Additionally, this exon contributed to changes of gray matter volumes in multiple brain regions, including the visual cortex known to be involved in AUD. In conclusion, this study provides strong evidence that RNA alternative splicing impacts the susceptibility to AUD and adds new information on AUD-relevant genes and pathways. Our framework is also applicable to other types of splicing events and to other complex genetic disorders.
酒精使用障碍(AUD)是一种复杂的遗传性疾病,其特征是过度饮酒引发的各种问题。识别导致AUD风险的功能性基因变异是一个主要目标。RNA的可变剪接介导了从DNA到基因表达的遗传信息流,并扩展了蛋白质组的多样性。我们研究了可变剪接是否可能是AUD的一个风险因素。在此,我们采用基于孟德尔随机化(MR)的方法来识别导致AUD风险的外显子跳跃(大脑中主要的剪接事件)。来自CommonMind联盟的基因型和RNA测序数据被用作训练数据集,以建立将个体基因型与前额叶皮质中外显子跳跃相联系的预测模型。我们将这些模型应用于酒精中毒遗传学合作研究的数据,以检验推断的顺式调控剪接结果与AUD相关性状之间的关联。我们确定了27个预计会影响AUD风险的外显子跳跃事件;其中6个在澳大利亚酒精使用障碍双生子家族研究中得到了重复验证。它们的宿主基因是DRC1、ELOVL7、LINC00665、NSUN4、SRRM2和TBC1D5。这些剪接事件下游的基因在神经免疫途径中富集。在另外四项大规模全基因组关联研究中,ELOVL7外显子跳跃对AUD风险的MR推断影响得到了进一步支持。此外,这个外显子导致了多个脑区灰质体积的变化,包括已知与AUD有关的视觉皮质。总之,本研究提供了强有力的证据,表明RNA可变剪接会影响对AUD的易感性,并为与AUD相关的基因和途径增添了新信息。我们的框架也适用于其他类型的剪接事件和其他复杂的遗传性疾病。