Board P, Nishida T, Gatmaitan Z, Che M, Arias I M
Department of Physiology, Tufts University School of Medicine, Boston, Massachusetts 02111.
Hepatology. 1992 Apr;15(4):722-5. doi: 10.1002/hep.1840150428.
The Dubin-Johnson syndrome is manifested by conjugated hyperbilirubinemia and pigment accumulation in hepatocellular lysosomes. The TR-rat model is a phenotypic model of the Dubin-Johnson syndrome and is characterized by defective ATP-dependent transport of a group of nonbile acid organic anions, including glutathione-S-conjugates and oxidized glutathione, across the bile canaliculus. Similar ATP-dependent transport mechanisms have been described in erythrocytes. Intact erythrocytes and inverted erythrocyte membrane vesicles from Dubin-Johnson patients, TR-rats and appropriate controls were studied with regard to ATP-dependent transport of dinitrophenyl glutathione and oxidized glutathione. No significant differences were observed, indicating that the erythrocyte and canalicular ATP-dependent transporters for these substrates are functionally and potentially genetically distinct.
杜宾-约翰逊综合征表现为结合性高胆红素血症以及肝细胞溶酶体中的色素蓄积。TR大鼠模型是杜宾-约翰逊综合征的一种表型模型,其特征是一组非胆汁酸有机阴离子(包括谷胱甘肽-S-共轭物和氧化型谷胱甘肽)跨胆小管的ATP依赖性转运存在缺陷。在红细胞中也描述了类似的ATP依赖性转运机制。对来自杜宾-约翰逊综合征患者、TR大鼠和适当对照的完整红细胞及倒置红细胞膜囊泡进行了二硝基苯基谷胱甘肽和氧化型谷胱甘肽的ATP依赖性转运研究。未观察到显著差异,表明这些底物的红细胞和胆小管ATP依赖性转运体在功能上以及潜在的基因上是不同的。