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SOX7:通过多组学数据分析鉴定出的自闭症相关基因。

SOX7: Autism associated gene identified by analysis of multi-Omics data.

作者信息

Gonzales Samantha, Zhao Jane Zizhen, Choi Na Young, Acharya Prabha, Jeong Sehoon, Wang Xuexia, Lee Moo-Yeal

机构信息

Department of Biostatistics, Florida International University, Miami, Florida, United States of America.

Department of Psychology and Neuroscience, University of North Carolina at Chapel Hill, Chapel Hill, North Carolina, United States of America.

出版信息

PLoS One. 2025 May 15;20(5):e0320096. doi: 10.1371/journal.pone.0320096. eCollection 2025.

Abstract

Genome-wide association studies and next generation sequencing data analyses based on DNA information have identified thousands of mutations associated with autism spectrum disorder (ASD). However, more than 99% of identified mutations are non-coding. Thus, it is unclear which of these mutations might be functional and thus potentially causal variants. Transcriptomic profiling using total RNA-sequencing has been one of the most utilized approaches to link protein levels to genetic information at the molecular level. The transcriptome captures molecular genomic complexity that the DNA sequence solely does not. Some mutations alter a gene's DNA sequence but do not necessarily change expression and/or protein function. To date, few common variants reliably associated with the diagnosis status of ASD despite consistently high estimates of heritability. In addition, reliable biomarkers used to diagnose ASD or molecular mechanisms to define the severity of ASD do not exist. Therefore, it is necessary to integrate DNA and RNA testing together to identify true causal genes and propose useful biomarkers for ASD. We performed gene-based association studies with adaptive test using genome-wide association studies' (GWAS) summary statistics with two large GWAS datasets (ASD 2019 data: 18,382 ASD cases and 27,969 controls [discovery data]; ASD 2017 data: 6,197 ASD cases and 7,377 controls [replication data]) which were obtained from the Psychiatric Genomics Consortium (PGC). In addition, we investigated differential expression between ASD cases and controls for genes identified in gene-based GWAS with two RNA-seq datasets (GSE211154: 20 cases and 19 controls; GSE30573: 3 cases and 3 controls). We identified 5 genes significantly associated with ASD in ASD 2019 data (KIZ-AS1, p = 8.67 × 10-10; KIZ, p = 1.16 × 10-9; XRN2, p = 7.73 × 10-9; SOX7, p = 2.22 × 10-7; LOC101929229 also known as PINX1-DT, p = 2.14 × 10-6). Among these 5 genes, gene SOX7 (p = 0.00087) and LOC101929229 (p = 0.009) were replicated in ASD 2017 data. KIZ-AS1 (p = 0.059) and KIZ (p = 0.06) were close to the boundary of replication in ASD 2017 data. Genes SOX7 (p = 0.036 in all samples; p = 0.044 in white samples) indicated significant expression differences between cases and controls in the GSE211154 RNA-seq data. Furthermore, gene SOX7 was upregulated in cases than in controls in the GSE30573 RNA-seq data (p = 0.0017; Benjamini-Hochberg adjusted p = 0.0085). SOX7 encodes a member of the SOX (SRY-related HMG-box) family of transcription factors pivotally contributing to determining of the cell fate and identity in many lineages. The encoded protein may act as a transcriptional regulator after forming a protein complex with other proteins leading to autism. Gene SOX7 in the transcription factor family could be associated with ASD. This finding may provide new diagnostic and therapeutic strategies for ASD.

摘要

基于DNA信息的全基因组关联研究和下一代测序数据分析已经确定了数千个与自闭症谱系障碍(ASD)相关的突变。然而,超过99%已确定的突变是非编码的。因此,尚不清楚这些突变中哪些可能具有功能,从而可能是因果变异。使用全转录组RNA测序进行转录组分析一直是在分子水平上将蛋白质水平与遗传信息联系起来最常用的方法之一。转录组捕捉到了DNA序列单独无法体现的分子基因组复杂性。一些突变会改变基因的DNA序列,但不一定会改变表达和/或蛋白质功能。迄今为止,尽管遗传度估计一直很高,但很少有常见变异与ASD的诊断状态可靠相关。此外,不存在用于诊断ASD的可靠生物标志物或用于定义ASD严重程度的分子机制。因此,有必要将DNA和RNA检测结合起来,以确定真正的因果基因,并为ASD提出有用的生物标志物。我们使用来自精神病基因组学联盟(PGC)的两个大型全基因组关联研究数据集(ASD 2019数据:18382例ASD病例和27969例对照[发现数据];ASD 2017数据:6197例ASD病例和7377例对照[复制数据])的全基因组关联研究(GWAS)汇总统计数据,进行了基于基因的适应性检验关联研究。此外,我们使用两个RNA测序数据集(GSE211154:20例病例和19例对照;GSE30573:3例病例和3例对照),研究了基于基因的GWAS中确定的基因在ASD病例和对照之间的差异表达。我们在ASD 2019数据中确定了5个与ASD显著相关的基因(KIZ-AS1,p = 8.67×10^-10;KIZ,p = 1.16×10^-9;XRN2,p = 7.73×10^-9;SOX7,p = 2.22×10^-7;LOC101929229,也称为PINX1-DT,p = 2.14×10^-6)。在这5个基因中,基因SOX7(p = 0.00087)和LOC101929229(p = 0.009)在ASD 2017数据中得到了复制。KIZ-AS1(p = 0.059)和KIZ(p = 0.06)在ASD 2017数据中接近复制边界。基因SOX7(在所有样本中p = 0.036;在白人样本中p = 0.044)在GSE211154 RNA测序数据中显示出病例和对照之间的显著表达差异。此外,在GSE30573 RNA测序数据中,基因SOX7在病例中比在对照中上调(p = 0.0017;Benjamini-Hochberg校正p = 0.0085)。SOX7编码SOX(SRY相关HMG盒)转录因子家族的一个成员,该家族在许多谱系中对决定细胞命运和身份起着关键作用。编码的蛋白质在与其他导致自闭症的蛋白质形成蛋白质复合物后可能作为转录调节因子发挥作用。转录因子家族中的基因SOX7可能与ASD相关。这一发现可能为ASD提供新的诊断和治疗策略。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/567e/12080844/e6ab29b6e8ae/pone.0320096.g001.jpg

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