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Mitochondrial DNA mutation analysis from exome sequencing-A more holistic approach in diagnostics of suspected mitochondrial disease.外显子组测序中的线粒体 DNA 突变分析——疑似线粒体疾病诊断中的更全面方法。
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本文引用的文献

1
MitoScape: A big-data, machine-learning platform for obtaining mitochondrial DNA from next-generation sequencing data.MitoScape:一个从下一代测序数据中获取线粒体 DNA 的大数据、机器学习平台。
PLoS Comput Biol. 2021 Nov 11;17(11):e1009594. doi: 10.1371/journal.pcbi.1009594. eCollection 2021 Nov.
2
Validation and clinical performance of a combined nuclear-mitochondrial next-generation sequencing and copy number variant analysis panel in a Canadian population.加拿大人群中联合核-线粒体下一代测序和拷贝数变异分析面板的验证和临床性能。
Am J Med Genet A. 2021 Feb;185(2):486-499. doi: 10.1002/ajmg.a.61998. Epub 2020 Dec 10.
3
MITO-FIND: A study in 390 patients to determine a diagnostic strategy for mitochondrial disease.MITO-FIND:一项针对 390 名患者的研究,旨在确定线粒体疾病的诊断策略。
Mol Genet Metab. 2020 Sep-Oct;131(1-2):66-82. doi: 10.1016/j.ymgme.2020.08.009. Epub 2020 Sep 18.
4
REDBot: Natural language process methods for clinical copy number variation reporting in prenatal and products of conception diagnosis.REDBot:用于产前和妊娠产物诊断中临床拷贝数变异报告的自然语言处理方法。
Mol Genet Genomic Med. 2020 Nov;8(11):e1488. doi: 10.1002/mgg3.1488. Epub 2020 Sep 22.
5
Specifications of the ACMG/AMP standards and guidelines for mitochondrial DNA variant interpretation.ACMG/AMP 标准和线粒体 DNA 变异解读指南的规范。
Hum Mutat. 2020 Dec;41(12):2028-2057. doi: 10.1002/humu.24107. Epub 2020 Nov 10.
6
The diagnostic utility of genome sequencing in a pediatric cohort with suspected mitochondrial disease.基因组测序在疑似线粒体疾病的儿科队列中的诊断效用。
Genet Med. 2020 Jul;22(7):1254-1261. doi: 10.1038/s41436-020-0793-6. Epub 2020 Apr 21.
7
Panel-Based Nuclear and Mitochondrial Next-Generation Sequencing Outcomes of an Ethnically Diverse Pediatric Patient Cohort with Mitochondrial Disease.基于panel 的核和线粒体新一代测序在一个具有线粒体疾病的多种族儿科患者队列中的结果。
J Mol Diagn. 2019 May;21(3):503-513. doi: 10.1016/j.jmoldx.2019.02.002. Epub 2019 Mar 11.
8
Mitochondrial and nuclear disease panel (Mito-aND-Panel): Combined sequencing of mitochondrial and nuclear DNA by a cost-effective and sensitive NGS-based method.线粒体与核疾病检测组合(Mito-aND-Panel):采用一种经济高效且灵敏的基于二代测序(NGS)的方法对线粒体DNA和核DNA进行联合测序。
Mol Genet Genomic Med. 2018 Nov;6(6):1188-1198. doi: 10.1002/mgg3.500. Epub 2018 Nov 8.
9
Whole Exome Sequencing Is the Preferred Strategy to Identify the Genetic Defect in Patients With a Probable or Possible Mitochondrial Cause.全外显子组测序是确定可能或疑似线粒体病因患者遗传缺陷的首选策略。
Front Genet. 2018 Oct 12;9:400. doi: 10.3389/fgene.2018.00400. eCollection 2018.
10
Updated recommendation for the benign stand-alone ACMG/AMP criterion.良性 ACMG/AMP 标准更新推荐
Hum Mutat. 2018 Nov;39(11):1525-1530. doi: 10.1002/humu.23642.

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.

DOI:10.1002/mgg3.1943
PMID:35388601
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC9184660/
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。