Kushner J P, Edwards C Q, Dadone M M, Skolnick M H
Gastroenterology. 1985 May;88(5 Pt 1):1232-8. doi: 10.1016/s0016-5085(85)80084-6.
Subnormal activity of hepatic uroporphyrinogen decarboxylase is responsible for the derangement of porphyrin biosynthesis in both sporadic and familial porphyria cutanea tarda, but the enzymatic defect is not clinically expressed in the absence of hepatic siderosis. The pedigree study described here offers support for the hypothesis that a single allele for HLA-linked hereditary hemochromatosis is responsible for the hepatic siderosis in sporadic porphyria cutanea tarda. A two-locus causation model for sporadic porphyria cutanea tarda might explain both the observed incidence of overt cases and the rarity of multiple affected individuals within a pedigree.
肝尿卟啉原脱羧酶活性降低是导致散发性和家族性迟发性皮肤卟啉病中卟啉生物合成紊乱的原因,但在没有肝铁沉积的情况下,这种酶缺陷不会在临床上表现出来。本文所述的家系研究支持了这样一种假说,即与HLA相关的遗传性血色素沉着症的单个等位基因是散发性迟发性皮肤卟啉病中肝铁沉积的原因。散发性迟发性皮肤卟啉病的双基因致病模型可能既解释了显性病例的观察发病率,也解释了家系中多个受累个体的罕见性。