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散发性包涵体肌炎中的多个线粒体DNA缺失:56例患者的研究

Multiple mitochondrial DNA deletions in sporadic inclusion body myositis: a study of 56 patients.

作者信息

Santorelli F M, Sciacco M, Tanji K, Shanske S, Vu T H, Golzi V, Griggs R C, Mendell J R, Hays A P, Bertorini T E, Pestronk A, Bonilla E, DiMauro S

机构信息

H. Houston Merritt Clinical Research Center for Muscular Dystrophy and Related Diseases--Department of Neurology, Columbia University, New York, NY, USA.

出版信息

Ann Neurol. 1996 Jun;39(6):789-95. doi: 10.1002/ana.410390615.

Abstract

Inclusion body myositis, a chronic inflammatory disorder, is the most common cause of myopathy in adults over the age of 50. Diagnosis is based on clinical features and distinctive morphological findings by both light and electron microscopy. The causes of inclusion body myositis are still unknown. Ultrastructural mitochondrial changes and ragged-red fibers are common in patients with sporadic inclusion body myositis, and multiple [correction of mutiple] mitochondrial DNA (mtDNA) deletions have been reported in 3 such patients, suggesting that mtDNA mutations may have a pathogenetic role. We studied 56 patients with sporadic inclusion body myositis, using a combination of clinical, morphological, biochemical, and molecular genetic analyses to determine the frequency and the distribution of mtDNA deletions. Using the polymerase chain reaction, we found multiple mtDNA deletions in 73% of patients, compared to 40% of normal age-matched control subjects and 47% of disease control subjects. The presence of deletions correlated with morphological evidence of ragged-red, cytochrome c oxidase-negative fibers, and with defects of complexes I and IV of the electron transport chain. Although aging may account for a proportion of mtDNA deletions in patients with sporadic inclusion body myositis and control subjects, mtDNA alterations may be accelerated in sporadic inclusion body myositis.

摘要

包涵体肌炎是一种慢性炎症性疾病,是50岁以上成年人肌病最常见的病因。诊断基于临床特征以及光镜和电镜下独特的形态学表现。包涵体肌炎的病因仍不清楚。超微结构线粒体改变和破碎红纤维在散发性包涵体肌炎患者中很常见,已有3例此类患者报告存在多个线粒体DNA(mtDNA)缺失,提示mtDNA突变可能具有致病作用。我们对56例散发性包涵体肌炎患者进行了研究,结合临床、形态学、生化和分子遗传学分析来确定mtDNA缺失的频率和分布。使用聚合酶链反应,我们发现73%的患者存在多个mtDNA缺失,而年龄匹配的正常对照受试者为40%,疾病对照受试者为47%。缺失的存在与破碎红、细胞色素c氧化酶阴性纤维的形态学证据以及电子传递链复合体I和IV的缺陷相关。虽然衰老可能在散发性包涵体肌炎患者和对照受试者的mtDNA缺失中占一定比例,但散发性包涵体肌炎患者的mtDNA改变可能会加速。

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