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PMID: 2118657 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, P.H.S.

Dysfunctional C1 inhibitor Ta: deletion of Lys-251 results in acquisition of an N-glycosylation site.

Parad RB, Kramer J, Strunk RC, Rosen FS, Davis AE

Abstract

Hereditary angioneurotic edema is inherited as an autosomal dominant disorder and is characterized by potentially life-threatening episodic angioedema. In type II hereditary angioneurotic edema, a dysfunctional C1 inhibitor molecule is present together with low levels of normal C1 inhibitor. About 70% of these dysfunctional proteins result from reactive center (Arg-444) mutations. We describe the deletion of nucleotides encoding Lys-251 (AAG) in C1 inhibitor Ta, the dysfunctional C1 inhibitor from a family with type II hereditary angioneurotic edema. DNA sequence analysis was derived from clones obtained through polymerase chain reaction amplification of blood monocyte C1 inhibitor mRNA. As expected, clones with both normal and abnormal sequence were isolated. The deletion was verified by protein sequence analysis. These data, together with biochemical analysis of the protein and cell-free translation studies, suggest that this deletion, by altering the normal amino acid sequence from Asn-Lys-Ile-Ser to Asn-Ile-Ser, creates a new glycosylation site. The additional carbohydrate accounts for the larger size on SDS/PAGE and very likely interferes with protein function.

MeSH Terms
Amino Acid Sequence Angioedema/genetics Base Sequence Cell-Free System Chromosome Deletion Complement C1 Inactivator Proteins/genetics,isolation & purification Electrophoresis, Polyacrylamide Gel Glycosylation Humans Lysine Molecular Sequence Data Molecular Weight Protein Biosynthesis RNA, Messenger/genetics
Chemicals
Complement C1 Inactivator Proteins RNA, Messenger complement C1 inhibitor Ta Lysine
Authors & Affiliations
5 authors, click to expand affiliations / ORCID
Parad R B
Department of Pediatrics, Harvard Medical School, Boston, MA.
Kramer J
Strunk R C
Rosen F S
Davis A E
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34 references, click to expand
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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
1990-09-00
Pages
6786-90
Language
English
Region
United States
NLM ID
7505876
PMCID
PMC54622
Subset
IM
Grants
NIAID NIH HHS · AI23401 · United States
NICHD NIH HHS · HD17461 · United States
NICHD NIH HHS · HD22082 · United States
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