Gitlin J D
Edward Mallinckrodt Department of Pediatrics, Washington University School of Medicine, St. Louis, Missouri 63130, USA.
Pediatr Res. 1998 Sep;44(3):271-6. doi: 10.1203/00006450-199809000-00001.
Aceruloplasminemia is an autosomal recessive disorder of iron metabolism characterized by diabetes, retinal degeneration, and neurologic symptoms. Affected patients evidence marked parenchymal iron accumulation in conjunction with an absence of circulating serum ceruloplasmin and molecular genetic analysis reveals inherited mutations in the ceruloplasmin gene. Taken together with earlier studies that characterized ceruloplasmin as a ferroxidase and recent work indicating an essential role for a homologous multicopper oxidase in iron metabolism in Saccharomyces cerevisiae, these findings reveal an essential role for ceruloplasmin in human iron metabolism. The presence of neurologic symptoms in patients with aceruloplasminemia is unique among the characterized disorders of iron metabolism, and recent findings indicate that astrocyte-specific ceruloplasmin gene expression is critical for iron metabolism and neuronal survival in the retina and basal ganglia. The discovery of this disease provides new insights into the pathways of CNS iron metabolism of direct relevance to a variety of nutritional and genetic disorders of childhood.
无铜蓝蛋白血症是一种常染色体隐性铁代谢紊乱疾病,其特征为糖尿病、视网膜变性和神经症状。受影响的患者表现出明显的实质铁蓄积,同时循环血清铜蓝蛋白缺失,分子遗传学分析显示铜蓝蛋白基因存在遗传性突变。结合早期将铜蓝蛋白表征为铁氧化酶的研究以及最近表明同源多铜氧化酶在酿酒酵母铁代谢中起关键作用的工作,这些发现揭示了铜蓝蛋白在人类铁代谢中的关键作用。无铜蓝蛋白血症患者出现神经症状在已表征的铁代谢紊乱疾病中是独特的,最近的发现表明星形胶质细胞特异性铜蓝蛋白基因表达对于视网膜和基底神经节的铁代谢及神经元存活至关重要。这种疾病的发现为中枢神经系统铁代谢途径提供了新见解,这与多种儿童营养和遗传疾病直接相关。