Inui K, Fukushima H, Tsukamoto H, Taniike M, Midorikawa M, Tanaka J, Nishigaki T, Okada S
Department of Pediatrics, Osaka University School of Medicine, Japan.
J Pediatr. 1992 Jan;120(1):62-6. doi: 10.1016/s0022-3476(05)80599-2.
Four families with mitochondrial encephalomyopathy are described. Probands of three families had typical clinical presentations of mitochondrial myopathy, encephalopathy, lactic acidosis, and strokelike episodes (MELAS), but the proband of family 4 lacked strokelike episodes. The mitochondrial DNA mutation of tRNA(Leu(UUR)) (transfer ribonucleic acid specific to leucine (UUR codon)) found in MELAS was examined in muscle DNA obtained from biopsy samples of the probands of four families and the maternal relatives of family 2. The mutation was detected in all muscle samples, and the degree of the mutated DNA was 68% to 84% by Southern blot analysis. However, the clinical patterns of the maternal relatives of family 2 were mild and distinctly different from MELAS. The same mutation was also detected in blood-derived DNA samples of all family members examined, including healthy mothers but not fathers, although the degree of mutation did not correlate with the clinical severity. These results confirmed the maternal inheritance of this disease and suggested that the mitochondrial DNA mutation (tRNA(Leu(UUR))) may cause clinical symptoms other than MELAS. The clinical findings of mitochondrial encephalomyopathy should be reinvestigated in terms of the mitochondrial gene mutation; the polymerase chain reaction method will be useful for screening for this mutation of mitochondrial DNA in blood samples.
本文描述了四个患有线粒体脑肌病的家庭。三个家庭的先证者具有线粒体肌病、脑病、乳酸性酸中毒和类卒中发作(MELAS)的典型临床表现,但家庭4的先证者没有类卒中发作。对四个家庭先证者的活检肌肉样本以及家庭2的母系亲属的肌肉DNA,检测了MELAS中发现的线粒体DNA tRNA(Leu(UUR))(亮氨酸特异性转运核糖核酸(UUR密码子))突变。在所有肌肉样本中均检测到该突变,通过Southern印迹分析,突变DNA的比例为68%至84%。然而,家庭2的母系亲属的临床症状较轻,与MELAS明显不同。在所有检测的家庭成员的血液来源DNA样本中也检测到相同的突变,包括健康的母亲但不包括父亲,尽管突变程度与临床严重程度无关。这些结果证实了该疾病的母系遗传,并提示线粒体DNA突变(tRNA(Leu(UUR)))可能导致除MELAS之外的临床症状。应根据线粒体基因突变对线粒体脑肌病的临床发现进行重新研究;聚合酶链反应方法将有助于在血液样本中筛查这种线粒体DNA突变。