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PMID: 2243128 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't

Two different point G to A mutations in exon 10 of the porphobilinogen deaminase gene are responsible for acute intermittent porphyria.

The Journal of clinical investigation ·Vol. 86 ·No. 5 ·1990-11-00 ·Pages 1511-6

Delfau MH, Picat C, de Rooij FW, Hamer K, Bogard M, Wilson JH, Deybach JC, Nordmann Y, Grandchamp B

Abstract

Two mutations of the porphobilinogen (PBG) deaminase gene resulting in cross-reacting immunological material (CRIM) positive forms of acute intermittent porphyria (AIP) have been identified by in vitro amplification of cDNA and cloning of the amplified products in a bacterial expression vector. Both mutations resulted from G to A transitions in exon 10 of the gene and produced arginine to glutamine substitutions in the abnormal protein. Expression of mutant cDNA in Escherichia coli reveals that one but not the other of these amino acid changes results in a striking decrease of the optimal pH of the mutated enzyme. One or the other of these two mutations accounted for the defect causing AIP in six unrelated patients among the eight patients evaluated with the CRIM positive subtype of this disorder.

MeSH Terms
Acute Disease Amino Acid Sequence Base Sequence Blotting, Western Cloning, Molecular Escherichia coli/genetics Exons Genes Humans Hydrogen-Ion Concentration Hydroxymethylbilane Synthase/genetics Molecular Sequence Data Mutation Polymerase Chain Reaction Porphyrias/enzymology,genetics
Chemicals
Hydroxymethylbilane Synthase
Authors & Affiliations
9 authors, click to expand affiliations / ORCID
Delfau M H
Laboratoire de Génétique Moleculaire, Faculté Xavier Bichat, Paris, France.
Picat C
de Rooij F W
Hamer K
Bogard M
Wilson J H
Deybach J C
Nordmann Y
Grandchamp B
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18 references, click to expand
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Article Info
Journal
The Journal of clinical investigation
Abbr.
J Clin Invest
ISSN
0021-9738
Published
1990-11-00
Pages
1511-6
Language
English
Region
United States
NLM ID
7802877
PMCID
PMC296897
Subset
IM
Databases
GENBANK
M60888
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