Hepatoerythropoietic porphyria: a new uroporphyrinogen decarboxylase defect or homozygous porphyria cutanea tarda?

Lancet. 1981 Apr 25;1(8226):916-9. doi: 10.1016/s0140-6736(81)91615-9.

Abstract

Uroporphyrinogen decarboxylase levels were measured in haemolysed whole blood or fibroblasts from 3 unrelated patients with hepatoerythropoietic porphyria (HEP) and in 4 unrelated patients with familial porphyria cutanea tarda, a condition in which the enzyme is defective. In HEP patients enzyme activities were 7% of normal in erythrocytes and 8% of normal in cultured skin fibroblasts. All the features of HEP, including the characteristic accumulation of protoporphyrin in erythrocytes, are secondary to this enzyme defect. The father of 1 HEP patient was heterozygous for the same enzyme defect. He also had uroporphyrinuria and was therefore indistinguishable from patients with subclinical familial porphyria cutanea tarda. It is suggested that patients with HEP are homozygous for the gene that causes porphyria cutanea tarda.

Publication types

  • Case Reports
  • Comparative Study

MeSH terms

  • Child
  • Erythrocytes / enzymology
  • Fibroblasts / metabolism
  • Homozygote
  • Humans
  • Liver / enzymology
  • Male
  • Middle Aged
  • Porphyrias / etiology*
  • Porphyrias / genetics
  • Porphyrins / metabolism
  • Skin Diseases / etiology*
  • Skin Diseases / genetics
  • Uroporphyrinogen Decarboxylase / deficiency
  • Uroporphyrinogen Decarboxylase / metabolism

Substances

  • Porphyrins
  • Uroporphyrinogen Decarboxylase