Akman Cigdem Inan, Sue Carolyn M, Shanske Sara, Tanji Kurenai, Bonilla Eduardo, Ojaimi Joceline, Krishna Sindu, Schubert Romaine, DiMauro Salvatore
Department of Neurology, Columbia University College of Physicians and Surgeons, New York, NY 10032, USA.
J Child Neurol. 2004 Apr;19(4):258-61. doi: 10.1177/088307380401900403.
A 3 1/2-year-old boy presented with megaloblastic anemia and recurrent episodes of severe lactic acidosis and coma. At age 4 years, he developed sepsis and died; postmortem examination failed to show any gross abnormality in any tissue. Biochemical analysis of muscle showed decreased activities for all respiratory chain enzymes except complex II. Muscle histochemistry revealed diffuse cytochrome c oxidase deficiency. Southern blot analysis of mitochondrial DNA from muscle, liver, and blood showed a heteroplasmic single mitochindrial DNA deletion of 2.4 kb, which removed the genes for cytochrome c oxidase I and II and the transfer ribonucleic acid genes for serine and aspartic acid. Single large-scale deletions in mitochondrial DNA have been associated with Pearson's syndrome, Kearns-Sayre syndrome, and progressive external ophthalmoplegia. This patient's presentation is unusual and suggests an overlap between Pearson's syndrome and Kearns-Sayre syndrome.
一名3岁半的男孩出现巨幼细胞贫血以及反复发作的严重乳酸酸中毒和昏迷。4岁时,他患上败血症并死亡;尸检未发现任何组织有明显异常。肌肉生化分析显示,除了复合物II外,所有呼吸链酶的活性均降低。肌肉组织化学显示细胞色素c氧化酶弥漫性缺乏。对肌肉、肝脏和血液中的线粒体DNA进行Southern印迹分析,发现一个2.4 kb的异质性单一线粒体DNA缺失,该缺失去除了细胞色素c氧化酶I和II的基因以及丝氨酸和天冬氨酸的转运核糖核酸基因。线粒体DNA中的单一大规模缺失与皮尔逊综合征、卡恩斯-塞尔综合征和进行性眼外肌麻痹有关。该患者的表现不寻常,提示皮尔逊综合征和卡恩斯-塞尔综合征之间存在重叠。