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全外显子组测序揭示脑动静脉畸形中的致病性 SIRT1 变异:病例报告。

Whole-Exome Sequencing Reveals Pathogenic SIRT1 Variant in Brain Arteriovenous Malformation: A Case Report.

机构信息

National Center for Biotechnology, 13/5 Kurgalzhynskoye Road, Astana 010000, Kazakhstan.

Laboratory of Bioinformatics and Systems Biology, Center for Life Science, National Laboratory Astana, Nazarbayev University, 53 Kabanbay Batyr Ave., Astana 010000, Kazakhstan.

出版信息

Genes (Basel). 2022 Sep 21;13(10):1689. doi: 10.3390/genes13101689.

Abstract

Arteriovenous malformations of the brain (bAVMs) are plexuses of pathological arteries and veins that lack a normal capillary system between them. Intracranial hemorrhage (hemorrhagic stroke) is the most frequent clinical manifestation of AVM, leading to lethal outcomes that are especially high among children and young people. Recently, high-throughput genome sequencing methods have made a notable contribution to the research progress in this subject. In particular, whole-exome sequencing (WES) methods allow the identification of novel mutations. However, the genetic mechanism causing AVM is still unclear. Therefore, the aim of this study was to investigate the potential genetic mechanism underlying AVM. We analyzed the WES data of blood and tissue samples of a 30-year-old Central Asian male diagnosed with AVM. We identified 54 polymorphisms in 43 genes. After in-silica overrepresentation enrichment analysis of the polymorphisms, the SIRT1 gene variant (g.67884831C>T) indicated a possible molecular mechanism of bAVM. Further studies are required to evaluate the functional impact of SIRT1 g.67884831C>T, which may warrant further replication and biological investigations related to sporadic bAVM.

摘要

脑动静脉畸形(bAVMs)是由病理动脉和静脉组成的丛,其间缺乏正常的毛细血管系统。颅内出血(出血性中风)是 AVM 最常见的临床表现,导致儿童和年轻人的致死率特别高。最近,高通量基因组测序方法为这一课题的研究进展做出了显著贡献。特别是全外显子组测序(WES)方法可以识别新的突变。然而,导致 AVM 的遗传机制仍不清楚。因此,本研究旨在探讨 AVM 的潜在遗传机制。我们分析了一名 30 岁中亚男性 AVM 患者的血液和组织样本的 WES 数据。我们在 43 个基因中发现了 54 个多态性。对多态性进行计算机过度表现富集分析后,SIRT1 基因变异(g.67884831C>T)提示了 bAVM 的可能分子机制。需要进一步研究来评估 SIRT1 g.67884831C>T 的功能影响,这可能需要进一步复制和进行与散发性 bAVM 相关的生物学研究。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/aeb1/9601721/4615f0f2785c/genes-13-01689-g001.jpg

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