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由TK2基因突变引起的肌病性线粒体DNA耗竭综合征

Myopathic mtDNA Depletion Syndrome Due to Mutation in TK2 Gene.

作者信息

Martín-Hernández Elena, García-Silva María Teresa, Quijada-Fraile Pilar, Rodríguez-García María Elena, Rivera Henry, Hernández-Laín Aurelio, Coca-Robinot David, Fernández-Toral Joaquín, Arenas Joaquín, Martín Miguel A, Martínez-Azorín Francisco

机构信息

1 Unidad Pediátrica de Enfermedades Raras, Metabólicas-Hereditarias y Mitocondriales, Departamento de Pediatría, Hospital Universitario 12 de Octubre, Madrid, Spain.

2 Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER), Madrid, Spain.

出版信息

Pediatr Dev Pathol. 2017 Sep-Oct;20(5):416-420. doi: 10.1177/1093526616686439. Epub 2017 Jan 25.

Abstract

Whole-exome sequencing was used to identify the disease gene(s) in a Spanish girl with failure to thrive, muscle weakness, mild facial weakness, elevated creatine kinase, deficiency of mitochondrial complex III and depletion of mtDNA. With whole-exome sequencing data, it was possible to get the whole mtDNA sequencing and discard any pathogenic variant in this genome. The analysis of whole exome uncovered a homozygous pathogenic mutation in thymidine kinase 2 gene ( TK2; NM_004614.4:c.323 C>T, p.T108M). TK2 mutations have been identified mainly in patients with the myopathic form of mtDNA depletion syndromes. This patient presents an atypical TK2-related myopathic form of mtDNA depletion syndromes, because despite having a very low content of mtDNA (<20%), she presents a slower and less severe evolution of the disease. In conclusion, our data confirm the role of TK2 gene in mtDNA depletion syndromes and expanded the phenotypic spectrum.

摘要

对一名患有发育不良、肌肉无力、轻度面部无力、肌酸激酶升高、线粒体复合物III缺乏和线粒体DNA(mtDNA)耗竭的西班牙女孩进行了全外显子组测序,以鉴定致病基因。利用全外显子组测序数据,可以获得完整的mtDNA测序结果,并排除该基因组中的任何致病变异。全外显子组分析发现胸苷激酶2基因(TK2;NM_004614.4:c.323 C>T,p.T108M)存在纯合致病突变。TK2突变主要在患有肌病形式的mtDNA耗竭综合征的患者中被发现。该患者呈现出一种非典型的与TK2相关的肌病形式的mtDNA耗竭综合征,因为尽管其mtDNA含量极低(<20%),但其疾病进展较慢且症状较轻。总之,我们的数据证实了TK2基因在mtDNA耗竭综合征中的作用,并扩大了其表型谱。

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