Restriction fragment length polymorphism analysis of the C1-inhibitor gene in hereditary C1-inhibitor deficiency

Clin Genet. 1991 Mar;39(3):161-71. doi: 10.1111/j.1399-0004.1991.tb03006.x.

Abstract

Four out of 12 kindreds with Type I hereditary angio-oedema (HAE) were shown to have unique disease-related restriction fragment length polymorphism (RFLPs) in one allele of the C1-inhibitor gene. These RFLPs were used to localise the gene mutations responsible for them in each family. The four mutations affected exon 4, exon 6, exon 7 and exon 8, respectively. Mutations in exon 6 and exon 8 have not been described previously in Type I HAE. The other two mutations which comprised an exon 4 deletion and an exon 7 deletion have already been documented by other investigators. In each family the mutation was seen to cosegregate with the disease. Detection of a disease-related RFLP in 30% of the Type I HAE kindred tested is higher than other published studies, and reflects the larger number of restriction enzymes employed. These results suggest that Type I HAE is likely to be associated with a multiplicity of gene mutations as is seen in other genetic diseases. A new C1-inhibitor gene-related RFLP in the normal population was also characterised. This may be useful as an indirect marker of the mutant C1-inhibitor allele in certain families with Type I HAE.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Angioedema / diagnosis
  • Angioedema / epidemiology
  • Angioedema / genetics*
  • Complement C1 Inactivator Proteins / deficiency*
  • Complement C1 Inactivator Proteins / genetics
  • DNA Mutational Analysis
  • DNA Probes
  • Exons
  • Genetic Diseases, Inborn / diagnosis
  • Genetic Diseases, Inborn / epidemiology
  • Genetic Diseases, Inborn / genetics*
  • Genetic Markers / genetics
  • Humans
  • Mutation / genetics*
  • Pedigree
  • Polymorphism, Genetic / genetics
  • Polymorphism, Restriction Fragment Length*
  • Restriction Mapping*

Substances

  • Complement C1 Inactivator Proteins
  • DNA Probes
  • Genetic Markers