Morten K J, Cooper J M, Brown G K, Lake B D, Pike D, Poulton J
Department of Paediatrics, University of Oxford, John Radcliffe Hospital, UK.
Hum Mol Genet. 1993 Dec;2(12):2081-7. doi: 10.1093/hmg/2.12.2081.
Point mutations in the mitochondrial gene tRNA leucine(UUR) have been associated with maternally inherited mitochondrial myopathies including the MELAS syndrome (Mitochondrial Myopathy Encephalopathy Lactic acidosis and Stroke-like episodes). We describe a further mutation in tRNA leucine(UUR) in a patient with mitochondrial encephalomyopathy, pigmentary retinopathy, dementia, hypoparathyroidism and diabetes mellitus. The mutation was heteroplasmic in the proband's blood (30%) and muscle (76%); it was present at high levels in the proband's affected mother (50% in muscle), and at low levels (< 10%) in blood, muscle and fibroblasts of an unaffected sister. The mutation was not found in 121 normal controls or 35 other patients with mitochondrial disorders. The mutation is at a highly conserved position in the tRNA molecule, close to the 3,243 mutation which is associated with more than 80% of MELAS cases. Further more, both mutations lie within a possible transcriptional control region. This finding adds further support to the evidence that mutations in this region and in other mitochondrial tRNA genes may cause disease.
线粒体基因亮氨酸转运RNA(UUR)中的点突变与母系遗传的线粒体肌病相关,包括MELAS综合征(线粒体肌病、脑病、乳酸酸中毒和卒中样发作)。我们描述了一名患有线粒体脑肌病、色素性视网膜病变、痴呆、甲状旁腺功能减退和糖尿病的患者中亮氨酸转运RNA(UUR)的另一种突变。该突变在先证者的血液(30%)和肌肉(76%)中为异质性;在先证者患病母亲的肌肉中以高水平存在(50%),而在未受影响的妹妹的血液、肌肉和成纤维细胞中以低水平存在(<10%)。在121名正常对照或35名其他线粒体疾病患者中未发现该突变。该突变位于转运RNA分子中一个高度保守的位置,靠近与80%以上的MELAS病例相关的3243突变。此外,这两种突变都位于一个可能的转录控制区域内。这一发现进一步支持了该区域及其他线粒体转运RNA基因突变可能导致疾病的证据。