基于回顾性分析的不明原因猝死易感基因的优先排序和降解模式表征。

IF 2.3 3区 医学 Q1 MEDICINE, LEGAL
Qi Shen, Zhimin Wang, Junyi Lin, Liliang Li, Suhua Zhang, Shouyu Wang, Chengtao Li
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引用次数: 0

摘要

已知遗传因素在表面健康个体的不明原因猝死(SUD)中起重要作用。目前,分子解剖被认为是SUD多学科治疗中有效的诊断工具。最近的研究强调了调控变异对复杂遗传疾病的贡献。在某些病例中,还发现了SUD易感基因非翻译区域内的致病变异。然而,编码区外致病变异的功能验证仍然具有挑战性。转录组分析是最直接的方法,可以与分子解剖同时进行,以识别SUD易感基因的异常表达,而心肌组织mRNA的死后降解使得转录组分析结果难以解释。在这项研究中,我们根据先前研究中病原遗传变异的分布进行了回顾性分析,并报道了分子尸检结果。在基因优先排序后,我们分析了基因型-组织表达数据库中432个不同采样时间间隔的左心室组织的转录组数据,以表征优先的SUD易感基因的降解模式。此外,研究了未冷冻和解冻样品的RNA降解差异。我们证明,根据基因的降解模式对其进行适当的分割,偏最小二乘判别分析可以有效识别正常样本和模拟SUD病例中目标基因的表达差异。综上所述,我们的研究结果为SUD法医调查期间RNA分析结果的解释提供了一种策略。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Retrospective analysis-based prioritization and degradation pattern characterization of sudden unexplained death susceptibility genes.

Genetic factors are known to have important roles in sudden unexplained death (SUD) of apparently healthy individuals. Currently, molecular autopsy is considered an effective diagnostic tool in the multidisciplinary management of SUD. Recent studies highlighted the contribution of regulatory variants to complex genetic disorders. Pathogenic variants within the untranslated regions of SUD susceptibility genes were also identified in certain cases. However, the functional validation of pathogenic variants outside of the coding regions remains challenging. As the most direct method, transcriptome analysis could be performed at the same time with molecular autopsy to identify the abnormal expression of SUD susceptibility genes, while the post-mortem degradation of mRNA in myocardial tissues has made it difficult to interpret the transcriptome profiling results. In this study, we performed a retrospective analysis-based prioritization of SUD susceptibility genes based on the distribution of pathogenic genetic variants in previous studies with molecular autopsy findings reported. After gene prioritization, we analyzed the transcriptome data of 432 left ventricle tissues with different sampling time intervals from the Genotype-Tissue Expression database, in order to characterize the degradation pattern of prioritized SUD susceptibility genes. Furthermore, RNA degradation difference between unfrozen and thawed samples was investigated. We demonstrated that with proper segmentation of genes according to their degradation patterns, a partial least squares-discriminant analysis could effectively recognize the expression difference of targeted genes between normal samples and simulated SUD cases. Taken together, our findings presented a strategy for the interpretation of RNA profiling results during the forensic investigation of SUD.

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来源期刊
CiteScore
5.80
自引率
9.50%
发文量
165
审稿时长
1 months
期刊介绍: The International Journal of Legal Medicine aims to improve the scientific resources used in the elucidation of crime and related forensic applications at a high level of evidential proof. The journal offers review articles tracing development in specific areas, with up-to-date analysis; original articles discussing significant recent research results; case reports describing interesting and exceptional examples; population data; letters to the editors; and technical notes, which appear in a section originally created for rapid publication of data in the dynamic field of DNA analysis.
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