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新生儿重症监护中快速串联线粒体基因组测序与外显子组测序对 Pearson 综合征的早期诊断

Early diagnosis of Pearson syndrome in neonatal intensive care following rapid mitochondrial genome sequencing in tandem with exome sequencing.

机构信息

Victorian Clinical Genetics Services, Murdoch Children's Research Institute, Melbourne, VIC, Australia.

Department of Paediatrics, University of Melbourne, Melbourne, VIC, Australia.

出版信息

Eur J Hum Genet. 2019 Dec;27(12):1821-1826. doi: 10.1038/s41431-019-0477-3. Epub 2019 Jul 29.

Abstract

Rapid genomic testing is a valuable new diagnostic tool for acutely unwell infants, however exome sequencing does not deliver clinical-grade mitochondrial genome sequencing and may fail to diagnose mitochondrial disorders caused by mitochondrial DNA (mtDNA) variants. Rapid mitochondrial genome sequencing and analysis are not routinely available in rapid genomic diagnosis programmes. We present two critically ill neonates with transfusion-dependent anaemia and persistent lactic acidosis who underwent rapid mitochondrial genome sequencing in tandem with exome sequencing as part of an exome sequencing-based rapid genomic diagnosis programme. No diagnostic variants were identified on examination of the nuclear exome data for either infant. Mitochondrial genome sequencing identified a large mtDNA deletion in both infants, diagnosing Pearson syndrome within 74 and 55 h, respectively. Early diagnosis in the third week of life allowed the avoidance of a range of other investigations and appropriate treatment planning. Rapid mitochondrial genome analysis provides additional diagnostic and clinical utility and should be considered as an adjunct to exome sequencing in rapid genomic diagnosis programmes.

摘要

快速基因组检测是一种有价值的急性不适婴儿新诊断工具,然而外显子组测序无法提供临床级别的线粒体基因组测序,并且可能无法诊断由线粒体 DNA(mtDNA)变异引起的线粒体疾病。快速线粒体基因组测序和分析在快速基因组诊断计划中通常不可用。我们介绍了两名严重贫血和持续乳酸酸中毒的危重新生儿,他们在进行外显子组测序的同时进行快速线粒体基因组测序,作为基于外显子组测序的快速基因组诊断计划的一部分。在对两个婴儿的核外显子数据进行检查时,均未发现诊断性变异。线粒体基因组测序在两名婴儿中均发现了大片段 mtDNA 缺失,分别在 74 小时和 55 小时内诊断为 Pearson 综合征。在生命的第三周进行早期诊断,可以避免一系列其他检查,并进行适当的治疗计划。快速线粒体基因组分析提供了额外的诊断和临床效用,应考虑将其作为快速基因组诊断计划中外显子组测序的辅助手段。

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