Fromenty B, Carrozzo R, Shanske S, Schon E A
Department of Neurology, College of Physicians and Surgeons, Columbia University, New York, New York 10032, USA.
Am J Med Genet. 1997 Sep 5;71(4):443-52. doi: 10.1002/(sici)1096-8628(19970905)71:4<443::aid-ajmg14>3.0.co;2-g.
Kearns-Sayre syndrome (KSS) is a sporadic multisystem mitochondrial disorder characterized by progressive external ophthalmoplegia, pigmentary retinopathy, onset before age 20, and severe cardiac conduction defects that can lead to death. KSS patients harbor partial deletions of mitochondrial DNA (delta-mtDNA), sometimes associated with the corresponding mtDNA duplication (dup-mtDNA). As reports on the distribution of dup-mtDNAs among KSS tissues are scarce, we searched for the presence of dup-mtDNAs in different autopsy tissues of two such patients, one of whom carried the so-called "common deletion." Using a newly developed long polymerase chain reaction (PCR) protocol in conjunction with Southern blot analyses, we found dup-mtDNAs in most of the examined tissues from both patients. The proportion of dup-mtDNA in these tissues was much lower than the proportion of delta-mtDNA, with one notable exception: in both patients, we found an unusually high level of dup-mtDNA in the heart. These data suggest that dup-mtDNAs may be more stable in heart tissue of KSS patients than in other long-lived postmitotic tissues.
卡恩斯-塞尔综合征(KSS)是一种散发性多系统线粒体疾病,其特征为进行性眼外肌麻痹、色素性视网膜病变、20岁前发病以及可导致死亡的严重心脏传导缺陷。KSS患者存在线粒体DNA(δ-mtDNA)的部分缺失,有时与相应的线粒体DNA重复(dup-mtDNA)相关。由于关于dup-mtDNA在KSS组织中分布的报道较少,我们在两名此类患者的不同尸检组织中寻找dup-mtDNA的存在,其中一名患者携带所谓的“常见缺失”。使用新开发的长聚合酶链反应(PCR)方案并结合Southern印迹分析,我们在两名患者的大多数检测组织中都发现了dup-mtDNA。这些组织中dup-mtDNA的比例远低于δ-mtDNA的比例,但有一个显著例外:在两名患者的心脏中,我们发现dup-mtDNA的水平异常高。这些数据表明,dup-mtDNA在KSS患者的心脏组织中可能比在其他长寿的有丝分裂后组织中更稳定。