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PMID: 63041 Published · ppublish English Journal Article

The primary enzyme defect in hereditary coproporphyria.

Lancet (London, England) ·Vol. 2 ·No. 7997 ·1976-12-04 ·Pages 1217-9

Elder GH, Evans JO, Thomas N

Abstract

The activity of coproporphyrinogen oxidase (E.C. 1.3.3.3) in cultured skin fibroblasts from three patients with hereditary coproporphyria (H.C.) was approximately half that in fibroblasts from normal subjects and patients with other types of porphyria. It is suggested that this is the primary defect in H.C., which is inherited as an autosomal dominant, and that the same abnormality is present in the liver. Consideration of the probable relative activities of the enzymes of haem biosynthesis in the liver in H.C. suggests that the acute attacks of porphyria which are its major clinical manifestation occur when the activity of uroporphyrinogen-I-synthase (E.C. 4.3.1.8) becomes rate-limiting for haem synthesis.

MeSH Terms
Adult Aged Chromosome Aberrations/complications Chromosome Disorders Coproporphyrinogens/urine Coproporphyrins/urine Feces/analysis Female Fibroblasts/enzymology Heme/biosynthesis Humans Hydroxymethylbilane Synthase/metabolism Liver/metabolism Male Oxidoreductases/deficiency Porphyrias/enzymology,metabolism,pathology Porphyrias, Hepatic Skin/pathology
Chemicals
Coproporphyrinogens Coproporphyrins Heme Oxidoreductases Hydroxymethylbilane Synthase
Authors & Affiliations
3 authors, click to expand affiliations / ORCID
Elder G H
Evans J O
Thomas N
Article Info
Journal
Lancet (London, England)
Abbr.
Lancet
ISSN
0140-6736
Published
1976-12-04
Pages
1217-9
Language
English
Region
England
NLM ID
2985213R
Subset
IM
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