Majamaa K, Moilanen J S, Uimonen S, Remes A M, Salmela P I, Kärppä M, Majamaa-Voltti K A, Rusanen H, Sorri M, Peuhkurinen K J, Hassinen I E
Department of Neurology, University of Oulu, Oulu, Finland.
Am J Hum Genet. 1998 Aug;63(2):447-54. doi: 10.1086/301959.
Mitochondrial diseases are characterized by considerable clinical variability and are most often caused by mutations in mtDNA. Because of the phenotypic variability, epidemiological studies of the frequency of these disorders have been difficult to perform. We studied the prevalence of the mtDNA mutation at nucleotide 3243 in an adult population of 245,201 individuals. This mutation is the most common molecular etiology of MELAS syndrome (mitochondrial encephalomyopathy, lactic acidosis, and strokelike episodes), one of the clinical entities among the mitochondrial disorders. Patients with diabetes mellitus, sensorineural hearing impairment, epilepsy, occipital brain infarct, ophthalmoplegia, cerebral white-matter disease, basal-ganglia calcifications, hypertrophic cardiomyopathy, or ataxia were ascertained on the basis of defined clinical criteria and family-history data. A total of 615 patients were identified, and 480 samples were examined for the mutation. The mutation was found in 11 pedigrees, and its frequency was calculated to be >=16. 3/100,000 in the adult population (95% confidence interval 11.3-21. 4/100,000). The mutation had arisen in the population at least nine times, as determined by mtDNA haplotyping. Clinical evaluation of the probands revealed a syndrome that most frequently consisted of hearing impairment, cognitive decline, and short stature. The high prevalence of the common MELAS mutation in the adult population suggests that mitochondrial disorders constitute one of the largest diagnostic categories of neurogenetic diseases.
线粒体疾病具有显著的临床变异性,最常见的病因是线粒体DNA(mtDNA)突变。由于表型变异性,对这些疾病发病率的流行病学研究很难开展。我们在245,201名成年人中研究了mtDNA第3243位核苷酸突变的患病率。该突变是线粒体脑肌病伴乳酸血症和卒中样发作(MELAS)综合征最常见的分子病因,MELAS综合征是线粒体疾病中的一种临床类型。根据明确的临床标准和家族史数据,确定患有糖尿病、感音神经性听力障碍、癫痫、枕叶脑梗死、眼肌麻痹、脑白质病、基底节钙化、肥厚型心肌病或共济失调的患者。共识别出615例患者,并对其中480份样本进行了该突变检测。在11个家系中发现了该突变,其在成年人群中的频率经计算≥16.3/100,000(95%置信区间为11.3 - 21.4/100,000)。通过mtDNA单倍型分析确定该突变在人群中至少出现过9次。对先证者的临床评估显示,最常见的综合征包括听力障碍、认知能力下降和身材矮小。成年人群中常见MELAS突变的高患病率表明,线粒体疾病是神经遗传性疾病中最大的诊断类别之一。