Literature DB >> 63041

The primary enzyme defect in hereditary coproporphyria.

G H Elder, J O Evans, N Thomas.   

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.

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Year:  1976        PMID: 63041     DOI: 10.1016/s0140-6736(76)91143-0

Source DB:  PubMed          Journal:  Lancet        ISSN: 0140-6736            Impact factor:   79.321


  17 in total

1.  Characterization of mutations in the CPO gene in British patients demonstrates absence of genotype-phenotype correlation and identifies relationship between hereditary coproporphyria and harderoporphyria.

Authors:  J Lamoril; H Puy; S D Whatley; C Martin; J R Woolf; V Da Silva; J C Deybach; G H Elder
Journal:  Am J Hum Genet       Date:  2001-04-16       Impact factor: 11.025

2.  Catecholamine uptake, accumulation, and release in acute porphyria.

Authors:  M F Beal; N O Atuk; T C Westfall; S M Turner
Journal:  J Clin Invest       Date:  1977-11       Impact factor: 14.808

3.  Chester porphyria: a clinical study of a new form of acute porphyria.

Authors:  M R Qadiri; S E Church; K E McColl; M R Moore; G R Youngs
Journal:  Br Med J (Clin Res Ed)       Date:  1986-02-15

4.  Neonatal-Onset Hereditary Coproporphyria: A New Variant of Hereditary Coproporphyria.

Authors:  Kosei Hasegawa; Hiroyuki Tanaka; Miho Yamashita; Yousuke Higuchi; Takayuki Miyai; Junko Yoshimoto; Ayumi Okada; Norihiro Suzuki; Keiji Iwatsuki; Hirokazu Tsukahara
Journal:  JIMD Rep       Date:  2017-03-28

5.  Coexistence of hereditary coproporphyria and epilepsy: coproporphyrinogen oxidase deficiency in liver and kidney.

Authors:  M Doss; R von Tiepermann; K H Pflüger
Journal:  J Neurol       Date:  1981       Impact factor: 4.849

6.  Hereditary coproporphyria: incidence in a large English family.

Authors:  J Andrews; H Erdjument; D C Nicholson
Journal:  J Med Genet       Date:  1984-10       Impact factor: 6.318

7.  Harderoporphyria: a variant hereditary coproporphyria.

Authors:  Y Nordmann; B Grandchamp; H de Verneuil; L Phung; B Cartigny; G Fontaine
Journal:  J Clin Invest       Date:  1983-09       Impact factor: 14.808

8.  Assignment of the human coproporphyrinogen oxidase to chromosome 9.

Authors:  B Grandchamp; D Weil; Y Nordmann; N Van Cong; H de Verneuil; C Foubert; M S Gross
Journal:  Hum Genet       Date:  1983       Impact factor: 4.132

9.  Allosteric inhibition of human lymphoblast and purified porphobilinogen deaminase by protoporphyrinogen and coproporphyrinogen. A possible mechanism for the acute attack of variegate porphyria.

Authors:  P Meissner; P Adams; R Kirsch
Journal:  J Clin Invest       Date:  1993-04       Impact factor: 14.808

10.  The "glucose effect" in acute hepatic porphyrias and in experimental porphyria.

Authors:  M Doss; F Verspohl
Journal:  Klin Wochenschr       Date:  1981-07-01
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