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线粒体脑肌病伴乳酸血症和卒中样发作(MELAS)中mtDNA A3243G突变负荷的纵向变化及功能水平

Longitudinal changes of mtDNA A3243G mutation load and level of functioning in MELAS.

作者信息

Mehrazin Mahsa, Shanske Sara, Kaufmann Petra, Wei Ying, Coku Jorida, Engelstad Kristin, Naini Ali, De Vivo Darryl C, DiMauro Salvatore

机构信息

Department of Neurology, Columbia University College of Physicians and Surgeons, New York, New York 10032, USA.

出版信息

Am J Med Genet A. 2009 Feb 15;149A(4):584-7. doi: 10.1002/ajmg.a.32703.

Abstract

Mitochondrial encephalopathy, lactic acidosis, and stroke-like episodes (MELAS), one of the most common mitochondrial multisystemic diseases, is most commonly associated with an A-to-G transition at nucleotide position 3243 (A3243G) in mitochondrial DNA. We studied 34 individuals harboring the A3243G mutation for up to 7 years; 17 had the full MELAS phenotype and 17 who were classified as "carrier relatives" because they were either asymptomatic or had some symptoms suggestive of mitochondrial disease but no seizures or strokes. Using the sensitive real-time polymerase chain reaction to quantify the A3243G mutation, we confirmed that the percent mutation decreases progressively in DNA isolated from blood: the average percent decrease was 0.5% per year for fully symptomatic patients and 0.2% per year for oligosymptomatic carrier relatives. We also correlated mutant loads with functional status estimated by the Karnofksky score: even though the mutation load decreases, the level of functioning worsens in fully symptomatic patients, whereas the level of functioning of carrier relatives remains largely unchanged. This study suggests that A3243G mutant load in DNA isolated from blood is neither useful for prognosis nor for functional assessment.

摘要

线粒体脑肌病伴乳酸血症和卒中样发作(MELAS)是最常见的线粒体多系统疾病之一,最常与线粒体DNA第3243位核苷酸(A3243G)的A到G转换相关。我们对34名携带A3243G突变的个体进行了长达7年的研究;其中17人具有典型的MELAS表型,另外17人被归类为“携带突变的亲属”,因为他们要么无症状,要么有一些提示线粒体疾病的症状,但没有癫痫发作或中风。使用灵敏的实时聚合酶链反应来定量A3243G突变,我们证实从血液中分离的DNA中突变百分比逐渐降低:完全有症状的患者每年平均降低0.5%,症状轻微的携带突变亲属每年平均降低0.2%。我们还将突变负荷与通过卡诺夫斯基评分估计的功能状态进行了关联:尽管突变负荷降低,但完全有症状的患者功能水平恶化,而携带突变亲属的功能水平基本保持不变。这项研究表明,从血液中分离的DNA中的A3243G突变负荷对预后和功能评估均无用处。

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