Mukerji S K, Pimstone N R
Biochem Biophys Res Commun. 1985 Mar 15;127(2):517-25. doi: 10.1016/s0006-291x(85)80190-x.
The pathogenesis of human porphyria cutanea tarda (PCT) is associated with an intrinsic abnormality of the uroporphyrinogen decarboxylase enzyme. To characterize this, we studied the kinetic properties of the red cell enzyme procured from patients with various forms of PCT and non-porphyric controls. The enzyme activity (units/mg hemoglobin) in the red cell hemolysate was close to normal in sporadic PCT but about 75% diminished in the familial PCT. The Michaelis constants (Km) of 200-fold purified red cell enzyme preparations, determined by using pentacarboxylic porphyrinogen I and uroporphyrinogen I as substrates, were more than 3.8-4.0 times higher, and the maximum velocity (Vmax) was about 70% diminished in familial PCT, whereas the Km was about 1.7-1.9 times higher and the Vmax was more or less normal for sporadic PCT. These observations suggest for the first time that the primary lesion in familial PCT is a genetically determined kinetic abnormality of uroporphyrinogen decarboxylase which appears to be different from the sporadic form of the disease.
迟发性皮肤卟啉症(PCT)的发病机制与尿卟啉原脱羧酶的内在异常有关。为对此进行表征,我们研究了从各种形式的PCT患者和非卟啉症对照者获取的红细胞酶的动力学特性。散发性PCT患者红细胞溶血产物中的酶活性(单位/毫克血红蛋白)接近正常,但家族性PCT患者中该酶活性降低约75%。以I型五羧基卟啉原和I型尿卟啉原为底物测定的200倍纯化红细胞酶制剂的米氏常数(Km)在家族性PCT中高出3.8 - 4.0倍以上,最大反应速度(Vmax)降低约70%,而散发性PCT中Km高出约1.7 - 1.9倍,Vmax基本正常。这些观察结果首次表明,家族性PCT的原发性病变是尿卟啉原脱羧酶的一种由基因决定的动力学异常,这似乎与散发性疾病形式不同。