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铜与铁的关联:遗传性无血浆铜蓝蛋白血症

The copper-iron connection: hereditary aceruloplasminemia.

作者信息

Nittis Thalia, Gitlin Jonathan D

机构信息

Edward Mallinckrodt Department of Pediatrics, Washington University School of Medicine, St Louis, MO 63110, USA.

出版信息

Semin Hematol. 2002 Oct;39(4):282-9. doi: 10.1053/shem.2002.35633.

Abstract

Hereditary aceruloplasminemia is an autosomal recessive disorder of iron homeostasis due to loss-of-function mutations in the ceruloplasmin gene. Affected individuals may present in adulthood with evidence of hepatic iron overload, diabetes, peripheral retinal degeneration, dystonia, dementia, or dysarthria. Laboratory studies demonstrate microcytic anemia, elevated serum ferritin, and a complete absence of serum ceruloplasmin ferroxidase activity. Consistent with the observed neurologic findings, magnetic resonance imaging reveals iron accumulation within the basal ganglia. Histologic studies detect abundant iron in hepatocytes, reticuloendothelial cells of the liver and spleen, beta cells of the pancreas, and astrocytes and neurons throughout the central nervous system. Characterization of this disorder reveals an essential role for ceruloplasmin in determining the rate of iron efflux from cells with mobilizable iron stores and provides new insights into the mechanisms of human iron metabolism.

摘要

遗传性血浆铜蓝蛋白缺乏症是一种常染色体隐性遗传性铁稳态失调疾病,由血浆铜蓝蛋白基因突变导致功能丧失引起。成年患者可能出现肝铁过载、糖尿病、周边视网膜变性、肌张力障碍、痴呆或构音障碍等症状。实验室检查显示小细胞性贫血、血清铁蛋白升高,且血清铜蓝蛋白铁氧化酶活性完全缺失。与观察到的神经学表现一致,磁共振成像显示基底神经节内有铁蓄积。组织学研究发现,肝细胞、肝脏和脾脏的网状内皮细胞、胰腺β细胞以及整个中枢神经系统的星形胶质细胞和神经元中都有大量铁沉积。对这种疾病的特征描述揭示了血浆铜蓝蛋白在决定可动员铁储存细胞中铁流出速率方面的重要作用,并为人类铁代谢机制提供了新的见解。

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