Kirches E J, Winkler K, Warich-Kirches M, Szibor R, Wien F, Kunz W S, von Bossanyi P, Bajaj P K, Dietzmann K
Institut für Neuropathologie, Universität Magdeburg, Germany.
J Inherit Metab Dis. 1998 Jun;21(4):400-8. doi: 10.1023/a:1005306725644.
The ratio of mtDNA and a nuclear reference gene was estimated by Southern blotting in the skeletal muscle DNA of a 3-year-old girl who suffered from congenital brain damage, focal epilepsy, hepatomegaly, malabsorption syndrome and severe myopathy. The signal ratio of mtDNA versus 18S rDNA was 22% of the mean value obtained from controls. No major deletions or insertions were found and the MERRF, MELAS and NARP mutations were ruled out. Mitochondrial DNA-encoded enzyme activities and mitochondrial respiration were reduced. The analysis of the NAD(P)H and flavoprotein redox states of intact fibres revealed the presence of mitochondrial dysfunction. In tissue sections a moderate elevation of type I and type II fibre diameter variation was detected, aberrant NADH- and succinate dehydrogenase staining and some ragged red fibres. This suggested that a mitochondrial disorder caused by a decrease in the amount of intact wild-type mtDNA was responsible for the severe myopathy.
通过Southern印迹法,对一名患有先天性脑损伤、局灶性癫痫、肝肿大、吸收不良综合征和严重肌病的3岁女孩的骨骼肌DNA中mtDNA与核参考基因的比例进行了估计。mtDNA与18S rDNA的信号比为对照组平均值的22%。未发现重大缺失或插入,排除了MERRF、MELAS和NARP突变。线粒体DNA编码的酶活性和线粒体呼吸降低。对完整纤维的NAD(P)H和黄素蛋白氧化还原状态分析显示存在线粒体功能障碍。在组织切片中,检测到I型和II型纤维直径变化适度升高、异常的NADH和琥珀酸脱氢酶染色以及一些破碎红纤维。这表明由完整野生型mtDNA数量减少引起的线粒体疾病是导致严重肌病的原因。