Bayat Ardeshir, Walter Joanne, Lambe Helen, Watson James S, Stanley John K, Marino Michael, Ferguson Mark W J, Ollier William E R
Center for Integrated Genomic Medical Research, University of Manchester, Manchester, United Kingdom.
Plast Reconstr Surg. 2005 Jan;115(1):134-41.
Dupuytren's disease is a familial fibroproliferative disorder of late onset affecting the hands. It is extremely common in individuals of Northern European extraction. Genetic studies have yet to identify the genes involved in the formation of the disease. Mitochondria play a critical role in cell metabolism and apoptosis. It is known that defective mitochondria generate abnormally high levels of reactive oxygen species by means of electron leak and that antioxidant enzyme activities decrease with age in skin fibroblasts. Respiratory function of mitochondria is also impaired in aging human tissues. Oxidative stress and production of free radicals may be important factors in the pathogenesis of Dupuytren's disease. Mitochondrial genes are also included in the regulation of apoptosis. Diseased tissue contains large numbers of myo- fibroblasts, which disappear by apoptosis during normal wound healing. High numbers of mitochondria have been observed in fibroblasts derived from diseased tissue. In the light of this evidence, the mitochondrial genome represents a potential location for candidate susceptibility genes for this late-onset disorder. In this study, the authors investigated the presence of mutations within the mitochondrial genome in 40 subjects; 20 Caucasian Dupuytren's disease patients with a maternally transmitted inheritance pattern and 20 control subjects were matched for age, sex, and race using a multiplex denaturing high-performance liquid chromatography approach. A hitherto unknown heteroplasmic mutation located within the mitochondrial 16s rRNA region was evident in 90 percent of patients and absent from all control subjects (p < 0.001; chi2 = 16.1). This mutation may be important in the pathogenesis of Dupuytren's disease.
掌腱膜挛缩症是一种迟发性的家族性纤维增生性疾病,会影响手部。在北欧后裔个体中极为常见。基因研究尚未确定该疾病形成过程中涉及的基因。线粒体在细胞代谢和细胞凋亡中起着关键作用。已知有缺陷的线粒体通过电子泄漏产生异常高水平的活性氧,并且抗氧化酶活性在皮肤成纤维细胞中会随着年龄增长而降低。在衰老的人体组织中线粒体的呼吸功能也会受损。氧化应激和自由基的产生可能是掌腱膜挛缩症发病机制中的重要因素。线粒体基因也参与细胞凋亡的调控。患病组织含有大量肌成纤维细胞,这些细胞在正常伤口愈合过程中会通过细胞凋亡而消失。在源自患病组织的成纤维细胞中观察到大量线粒体。鉴于这些证据,线粒体基因组是这种迟发性疾病候选易感基因的潜在定位点。在本研究中,作者使用多重变性高效液相色谱法,对40名受试者的线粒体基因组中的突变情况进行了调查;20名具有母系遗传模式的白种人掌腱膜挛缩症患者以及与患者年龄、性别和种族相匹配的20名对照受试者参与了研究。在90%的患者中发现了一个位于线粒体16s rRNA区域内的此前未知的异质性突变,而所有对照受试者中均未发现该突变(p < 0.001;卡方 = 16.)。这种突变可能在掌腱膜挛缩症的发病机制中具有重要意义