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全外显子组测序是诊断线粒体 DNA 疾病的一种替代方法。

Whole exome sequencing is an alternative method in the diagnosis of mitochondrial DNA diseases.

机构信息

Department of Neurology, Huashan Hospital, Fudan University, Shanghai, China.

Berry Genomics Co., Ltd, Beijing, China.

出版信息

Mol Genet Genomic Med. 2022 Jun;10(6):e1943. doi: 10.1002/mgg3.1943. Epub 2022 Apr 7.

Abstract

BACKGROUND

Mitochondrial disease (MD) is genetically a heterogeneous group of disorders with impairment in respiratory chain complexes or pathways associated with the mitochondrial function. Nowadays, it is still a challenge for the genetic screening of MD due to heteroplasmy of mitochondrial genome and the complex model of inheritance. This study was designed to investigate the feasibility of whole exome sequencing (WES)-based testing as an alternative option for the diagnosis of MD.

METHODS

A Chinese Han cohort of 48 patients with suspect MD features was tested using nanoWES, which was a self-designed WES technique that covered the complete mtDNA genome and 21,019 nuclear genes. Fourteen patients were identified with a single genetic variant and three with single deletion in mtDNA.

RESULTS

The heteroplasmy levels of variants in mitochondrial genome range from 11% to 100%. NanoWES failed to identify multiple deletions in mtDNA compared with long range PCR and massively parallel sequencing (LR-PCR/MPS). However, our testing showed obvious advantages in identifying variations in nuclear DNA. Based on nanoWES, we identified two patients with nuclear DNA variation. One of them showed Xp22.33-q28 duplication, which indicated a possibility of Klinefelter syndrome.

CONCLUSION

NanoWES yielded a diagnostic rate of 35.4% for MD. With the rapid advances of next generation sequencing technique and decrease in cost, we recommend the usage of nanoWES as a first-line method in clinical diagnosis.

摘要

背景

线粒体疾病(MD)是一组具有遗传异质性的疾病,其特征为呼吸链复合物或与线粒体功能相关的途径受损。由于线粒体基因组的异质性和复杂的遗传模式,目前对 MD 的基因筛查仍然是一个挑战。本研究旨在探讨基于全外显子组测序(WES)的检测作为 MD 诊断的替代方法的可行性。

方法

对 48 例具有 MD 特征的汉族患者进行了纳米 WES 检测,该技术是一种自行设计的 WES 技术,可覆盖完整的 mtDNA 基因组和 21019 个核基因。有 14 例患者发现单个基因突变,3 例患者发现 mtDNA 单缺失。

结果

线粒体基因组中变异的异质性水平从 11%到 100%不等。与长距离 PCR 和大规模平行测序(LR-PCR/MPS)相比,纳米 WES 无法识别 mtDNA 中的多个缺失。然而,我们的检测在核 DNA 变异的识别方面显示出明显的优势。基于纳米 WES,我们鉴定出了 2 例核 DNA 变异患者。其中 1 例患者存在 Xp22.33-q28 重复,提示可能患有 Klinefelter 综合征。

结论

纳米 WES 对 MD 的诊断率为 35.4%。随着下一代测序技术的快速发展和成本的降低,我们建议将纳米 WES 作为临床诊断的一线方法。

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