Ristow M, Giannakidou E, Hebinck J, Busch K, Vorgerd M, Kotzka J, Knebel B, Mueller-Berghaus J, Epplen C, Pfeiffer A, Kahn C R, Doria A, Krone W, Mueller-Wieland D
Klinik II und Poliklinik für Innere Medizin, Universität zu Köln, Cologne, Germany.
Diabetes. 1998 May;47(5):851-4. doi: 10.2337/diabetes.47.5.851.
Friedreich's ataxia is the most common hereditary ataxia and is frequently associated with disturbances of glucose metabolism. This autosomal recessive disease is caused by the decreased expression of a mitochondrial protein, frataxin, encoded by the X25 gene. Homozygous expansion of a GAA repeat in the first intron of X25 inhibits frataxin expression and is associated with clinical disease. We evaluated whether heterozygous expansions of the triplet repeat in the frataxin gene X25 may be associated with NIDDM in two genetically distinct populations--one in Germany (n = 358) and the other in the U.S. (n = 292)--using a polymerase chain reaction-based assay. Intermediate expansions (10-36 repeats), which are longer than normal but not sufficient for the appearance of the ataxia phenotype, were found in 24.7 and 27.3% of these two NIDDM cohorts compared with 7.6 and 6.3% of the matched control subjects (both P < 0.001). The odds ratios were 3.36 (95% CI 1.72-6.55) for the German group and 4.01 (2.08-7.74) for the U.S. group. Therefore, we conclude that the X25/frataxin GAA repeat polymorphism is associated with NIDDM in a frequency higher than any other mutation heretofore described. Further studies are needed to elucidate the possible role of frataxin in the pathogenesis of NIDDM.
弗里德赖希共济失调是最常见的遗传性共济失调,常与糖代谢紊乱相关。这种常染色体隐性疾病是由线粒体蛋白——由X25基因编码的铁调素表达减少所致。X25基因第一个内含子中GAA重复序列的纯合性扩增会抑制铁调素表达,并与临床疾病相关。我们使用基于聚合酶链反应的检测方法,在两个基因特征不同的人群中评估了铁调素基因X25中三联体重复序列的杂合性扩增是否与非胰岛素依赖型糖尿病(NIDDM)相关,其中一组来自德国(n = 358),另一组来自美国(n = 292)。在这两个NIDDM队列中,分别有24.7%和27.3%的患者存在中间扩增(10 - 36次重复),其长度长于正常水平,但不足以出现共济失调表型,而在匹配的对照组中这一比例分别为7.6%和6.3%(两者P均<0.001)。德国组的优势比为3.36(95%可信区间1.72 - 6.55),美国组为4.01(2.08 - 7.74)。因此,我们得出结论,X25/铁调素GAA重复多态性与NIDDM相关,其频率高于迄今所描述的任何其他突变。需要进一步研究以阐明铁调素在NIDDM发病机制中的可能作用。