Lambrecht Richard W, Bonkovsky Herbert L
Department of Medicine and Biochemistry, The Liver-Biliary-Pancreatic Center, University of Massachusetts Medical School, Worcester 01605-2324, USA.
Semin Gastrointest Dis. 2002 Apr;13(2):109-19.
A 52-year-old man presented to his primary care physician with blisters and sores on the backs of his hands. Laboratory studies supported a diagnosis of porphyria cutanea tarda, complicated by the presence of both the C282Y and H63D mutations in the HFE gene, with susequent iron over-load. This case illustrates the need to understand the pathogenesis of porphyria cutanea tarda, particularly the role of excess iron in the overproduction of uroporphyrin. Iron, by catalyzing the formation of reactive oxygen species, can enhance uroporphyrin formation by increasing the rate at which uroporphyrinogen is oxidized to urophophyrin. Iron may also act indirectly to inhibit uroporphyrinogen decarboxylase activity by enhancing the formation of non-porphyrin products of porphyrinogen oxidation that are themselves direct inhibitors of the enzyme. Finally, iron can act to increase urophorphyrin production by inducing delta-aminolevulinic acid synthase, thus increasing the amount of delta-aminolevulinic acid, the precursor to uroporphyrinogen, present in the cell. After the diagnosis, the patient underwent an aggressive series of therapeutic phlebotomies to reduce iron levels, and gradually the cutaneous manifestations of porphyria cutanea tarda improved.
一名52岁男性因手部背部出现水疱和溃疡而就诊于其初级保健医生。实验室检查支持迟发性皮肤卟啉症的诊断,该病症因HFE基因中同时存在C282Y和H63D突变并伴有铁过载而复杂化。该病例说明了了解迟发性皮肤卟啉症发病机制的必要性,尤其是过量铁在尿卟啉过度产生中的作用。铁通过催化活性氧的形成,可通过提高尿卟啉原氧化为尿卟啉的速率来增强尿卟啉的形成。铁还可能通过增强卟啉原氧化的非卟啉产物的形成而间接抑制尿卟啉原脱羧酶活性,这些非卟啉产物本身就是该酶的直接抑制剂。最后,铁可通过诱导δ-氨基-γ-酮戊酸合酶来增加尿卟啉的产生,从而增加细胞中尿卟啉原的前体δ-氨基-γ-酮戊酸的量。诊断后,患者接受了一系列积极的治疗性放血以降低铁水平,迟发性皮肤卟啉症的皮肤表现逐渐改善。