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RNA测序在肥厚型心肌病患者队列中用于单核苷酸变异鉴定的应用

Application of RNA-seq for single nucleotide variation identification in a cohort of patients with hypertrophic cardiomyopathy.

作者信息

Chumakova Anastasia, Vlasov Ivan, Filatova Elena, Klass Anna, Lysenko Andrey, Salagaev Gennady, Shadrina Maria, Slominsky Petr

机构信息

National Research Centre "Kurchatov Institute", Kurchatov sq. 2, Moscow, 123182, Russia.

Petrovsky National Research Center of Surgery, Abrikosovsky Ln 2, Moscow, 119991, Russia.

出版信息

Sci Rep. 2025 May 29;15(1):18788. doi: 10.1038/s41598-025-03226-x.

Abstract

A variety of techniques for DNA sequencing, such as specific gene sequencing, whole genome sequencing, or exome sequencing, are currently used to detect single nucleotide variations (SNVs). Although RNA-seq can be used to identify SNVs, studies that employ this approach are uncommon, and those that do often rely on outdated mapping methods or methods that are more suitable for genomic and exomic alignment. In this work, our aim is to apply modern RNA-seq specific alignment method in order to identify SNV in a cohort of HCMP patients, and characterize those SNV to gain insight into possible mechanisms of HCMP pathogenesis. The algorithm of identification of SNV based on transcriptomic sequencing data has been developed and evaluated. The algorithm was evaluated and the optimal quality threshold was determined based on allelic discrimination for the rs397516037 mutation (MYBPC3 c.3697 C > T) among patients. A total of 42,809 SNVs with a quality of 75 or higher were identified in 48 transcriptomes of hypertrophic cardiomyopathy (HCMP) myocardial tissue. Verification of missense and nonsense variants in key HCMP genes using Sanger sequencing confirmed the accuracy of the pipeline results. To identify variants potentially associated with HCMP pathogenesis, a filtration process was conducted based on minor allele frequency, substitution prediction score and ClinVar outcome. 214 missense mutations and 6 nonsense mutations were selected. Together with nonsense mutations, 19 mutations meeting the strictest SIFT and PolypPhen criteria were identified as potential factors influencing HCMP pathogenesis. We have developed and validated a method for identifying SNVs based on transcriptomic data, which can be used to identify putative pathogenic variants. We identified mutations in key HCMP genes MYBPC3 and MYH7 in a cohort of patients. We also found potentially pathologic mutations in genes ANXA6 and FEM1 A and obtained data supporting the role of NEBL in myocardial diseases. This method would be useful in analyzing transcriptomic data available in the Gene Expression Omnibus, but should be used with caution as we have tested it on a specific disease.

摘要

目前,多种DNA测序技术,如特定基因测序、全基因组测序或外显子组测序,被用于检测单核苷酸变异(SNV)。尽管RNA测序可用于识别SNV,但采用这种方法的研究并不常见,而且那些研究通常依赖过时的比对方法或更适合基因组和外显子组比对的方法。在这项工作中,我们的目标是应用现代RNA测序特异性比对方法,以便在一组肥厚型心肌病(HCMP)患者中识别SNV,并对这些SNV进行表征,以深入了解HCMP发病机制的可能机制。基于转录组测序数据识别SNV的算法已经开发并进行了评估。该算法经过评估,并根据患者中rs397516037突变(MYBPC3 c.3697 C>T)的等位基因鉴别确定了最佳质量阈值。在肥厚型心肌病(HCMP)心肌组织的48个转录组中,共鉴定出42,809个质量为75或更高的SNV。使用桑格测序法对关键HCMP基因中的错义变异和无义变异进行验证,证实了流程结果的准确性。为了识别可能与HCMP发病机制相关的变异,基于次要等位基因频率、替代预测分数和ClinVar结果进行了筛选过程。选择了214个错义突变和6个无义突变。连同无义突变一起,19个符合最严格SIFT和PolypPhen标准的突变被确定为影响HCMP发病机制的潜在因素。我们已经开发并验证了一种基于转录组数据识别SNV的方法,该方法可用于识别推定的致病变异。我们在一组患者中鉴定了关键HCMP基因MYBPC3和MYH7中的突变。我们还在基因ANXA6和FEM1 A中发现了潜在的病理性突变,并获得了支持NEBL在心肌疾病中作用的数据。这种方法在分析基因表达综合数据库中可用的转录组数据时将很有用,但由于我们仅在一种特定疾病上进行了测试,因此应谨慎使用。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d8b4/12122699/5cc7d7105eef/41598_2025_3226_Fig1_HTML.jpg

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