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PMID: 7937817 Published · ppublish English Journal Article Research Support, U.S. Gov't, P.H.S.

A cluster of mutations within a short triplet repeat in the C1 inhibitor gene.

Bissler JJ, Cicardi M, Donaldson VH, Gatenby PA, Rosen FS, Sheffer AL, Davis AE

Abstract

Mutations in the C1 inhibitor gene that result in low functional levels of C1 inhibitor protein cause hereditary angioneurotic edema. This disease is characterized by episodic edema leading to considerable morbidity and death. Among 60 unreported kindred with the disease, four patients were discovered to have mutations clustered within a 12-bp segment of exon 5 from nucleotide 8449 to nucleotide 8460. This short segment of DNA contains three direct repeats of the triplet CAA and is immediately preceded by a similar adenosine-rich sequence (CAAGAACAC). These triplet repeats make this region susceptible to mutation by a slipped mispairing mechanism. There are two other short triplet repeat elements in the coding region for this gene, but they have not become mutated in any kindred examined. This suggests that the apparent enhanced mutation rate in this region of exon 5 may be influenced by DNA structural characteristics.

MeSH Terms
Amino Acid Sequence Angioedema/blood,genetics Base Sequence Complement C1 Inactivator Proteins/genetics DNA/blood,isolation & purification DNA Primers DNA Transposable Elements Humans Leukocytes/metabolism Molecular Sequence Data Mutation Polymerase Chain Reaction Repetitive Sequences, Nucleic Acid Sequence Deletion
Chemicals
Complement C1 Inactivator Proteins DNA Primers DNA Transposable Elements DNA
Authors & Affiliations
7 authors, click to expand affiliations / ORCID
Bissler J J
Department of Pediatrics, University of Cincinnati, Children's Hospital Research Foundation, OH 45229.
Cicardi M
Donaldson V H
Gatenby P A
Rosen F S
Sheffer A L
Davis A E
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Article Info
Journal
Proceedings of the National Academy of Sciences of the United States of America
Abbr.
Proc Natl Acad Sci U S A
ISSN
0027-8424
Published
1994-09-27
Pages
9622-5
Language
English
Region
United States
NLM ID
7505876
PMCID
PMC44865
Subset
IM
Grants
NICHD NIH HHS · HD22082 · United States
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