Department of Biochemistry, University of Würzburg, Germany.
Wellcome Trust Sanger Institute, Hinxton, Cambridge, UK.
Nat Neurosci. 2013 Sep;16(9):1228-1237. doi: 10.1038/nn.3484. Epub 2013 Aug 4.
Implicating particular genes in the generation of complex brain and behavior phenotypes requires multiple lines of evidence. The rarity of most high-impact genetic variants typically precludes the possibility of accruing statistical evidence that they are associated with a given trait. We found that the enrichment of a rare chromosome 22q11.22 deletion in a recently expanded Northern Finnish sub-isolate enabled the detection of association between TOP3B and both schizophrenia and cognitive impairment. Biochemical analysis of TOP3β revealed that this topoisomerase was a component of cytosolic messenger ribonucleoproteins (mRNPs) and was catalytically active on RNA. The recruitment of TOP3β to mRNPs was independent of RNA cis-elements and was coupled to the co-recruitment of FMRP, the disease gene product in fragile X mental retardation syndrome. Our results indicate a previously unknown role for TOP3β in mRNA metabolism and suggest that it is involved in neurodevelopmental disorders.
要确定特定基因在复杂的大脑和行为表型的产生中的作用需要多种证据。大多数高影响力遗传变异的罕见性通常排除了积累它们与特定特征相关的统计证据的可能性。我们发现,在最近扩展的芬兰北部亚隔离人群中,22q11.22 号染色体罕见缺失的富集使得能够检测到 TOP3B 与精神分裂症和认知障碍之间的关联。TOP3β 的生化分析表明,这种拓扑异构酶是细胞质信使核糖核蛋白(mRNPs)的组成部分,并且对 RNA 具有催化活性。TOP3β 与 mRNPs 的募集与 RNA 顺式元件无关,并且与脆性 X 智力低下综合征的疾病基因产物 FMRP 的共同募集相关。我们的研究结果表明 TOP3β 在 mRNA 代谢中的一个先前未知的作用,并表明它参与了神经发育障碍。