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老派阿米什人群中的线粒体呼吸链疾病。

Mitochondrial respiratory chain disorders in the Old Order Amish population.

作者信息

Ghaloul-Gonzalez Lina, Goldstein Amy, Walsh Vockley Catherine, Dobrowolski Steven F, Biery Amy, Irani Afifa, Ibarra Jordan, Morton D Holmes, Mohsen Al-Walid, Vockley Jerry

机构信息

Department of Pediatrics, School of Medicine, University of Pittsburgh, Children's Hospital of Pittsburgh of UPMC, Pittsburgh, PA, USA.

Department of Pediatrics, School of Medicine, University of Pittsburgh, Children's Hospital of Pittsburgh of UPMC, Pittsburgh, PA, USA.

出版信息

Mol Genet Metab. 2016 Aug;118(4):296-303. doi: 10.1016/j.ymgme.2016.06.005. Epub 2016 Jun 16.

Abstract

The Old Order Amish populations in the US are one of the Plain People groups and are descendants of the Swiss Anabaptist immigrants who came to North America in the early eighteenth century. They live in numerous small endogamous demes that have resulted in reduced genetic diversity along with a high prevalence of specific genetic disorders, many of them autosomal recessive. Mitochondrial respiratory chain deficiencies arising from mitochondrial or nuclear DNA mutations have not previously been reported in the Plain populations. Here we present four different Amish families with mitochondrial respiratory chain disorders. Mutations in two mitochondrial encoded genes leading to mitochondrial respiratory chain disorder were identified in two patients. In the first case, MELAS syndrome caused by a mitochondrial DNA (mtDNA) mutation (m.3243A>G) was identified in an extended Amish pedigree following a presentation of metabolic strokes in the proband. Characterization of the extended family of the proband by a high resolution melting assay identified the same mutation in many previously undiagnosed family members with a wide range of clinical symptoms. A MELAS/Leigh syndrome phenotype caused by a mtDNA mutation [m.13513G>A; p.Asp393Asn] in the ND5 gene encoding the ND5 subunit of respiratory chain complex I was identified in a patient in a second family. Mutations in two nuclear encoded genes leading to mitochondrial respiratory chain disorder were also identified in two patients. One patient presented with Leigh syndrome and had a homozygous deletion in the NDUFAF2 gene, while the second patient had a homozygous mutation in the POLG gene, [c.1399G>A; p.Ala467Thr]. Our findings identify mitochondrial respiratory chain deficiency as a cause of disease in the Old Order Amish that must be considered in the context of otherwise unexplained systemic disease, especially if neuromuscular symptoms are present.

摘要

美国的旧秩序阿米什人群体是清教徒群体之一,是18世纪初来到北美地区的瑞士再洗礼派移民的后裔。他们生活在众多小型的族内通婚群落中,这导致了遗传多样性的降低以及特定遗传疾病的高发病率,其中许多是常染色体隐性遗传疾病。此前,在清教徒群体中尚未报道过由线粒体或核DNA突变引起的线粒体呼吸链缺陷。在此,我们报告了四个患有线粒体呼吸链疾病的不同阿米什家族。在两名患者中鉴定出两个线粒体编码基因的突变,这些突变导致了线粒体呼吸链疾病。在第一个病例中,在先证者出现代谢性中风后,在一个庞大的阿米什家系中鉴定出由线粒体DNA(mtDNA)突变(m.3243A>G)引起的线粒体脑肌病伴乳酸血症和卒中样发作(MELAS)综合征。通过高分辨率熔解分析对先证者的大家庭进行特征分析,发现在许多此前未被诊断出的有广泛临床症状的家庭成员中存在相同的突变。在第二个家族的一名患者中,鉴定出由编码呼吸链复合体I的ND5亚基的ND5基因中的mtDNA突变[m.13513G>A;p.Asp393Asn]引起的MELAS/ Leigh综合征表型。在另外两名患者中还鉴定出两个核编码基因的突变,这些突变导致了线粒体呼吸链疾病。一名患者表现为Leigh综合征,其NDUFAF2基因存在纯合缺失,而另一名患者的POLG基因存在纯合突变,[c.1399G>A;p.Ala467Thr]。我们的研究结果表明,线粒体呼吸链缺陷是旧秩序阿米什人群体中疾病的一个病因,在其他无法解释的全身性疾病的情况下,尤其是出现神经肌肉症状时,必须考虑到这一病因。

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