Acute intermittent porphyria: novel missense mutations in the human hydroxymethylbilane synthase gene

Genet Med. 2000 Sep-Oct;2(5):290-5. doi: 10.1097/00125817-200009000-00004.

Abstract

Purpose: To identify mutations in families with acute intermittent porphyria, an autosomal dominant inborn error of metabolism that results from the half-normal activity of the third enzyme in the heme biosynthetic pathway, hydroxymethylbilane synthase.

Methods: Mutations were identified by direct solid phase sequencing.

Results: Two novel missense mutations E80G and T78P and three previously reported mutations, R173W, G111R, and the splice site lesion, IVS1+1, were detected, each in an unrelated proband. The causality of the novel missense mutations was demonstrated by expression studies.

Conclusion: These findings provide for the precise diagnosis of carriers in these families and further expand the molecular heterogeneity of AIP.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Amino Acid Substitution*
  • Female
  • Gene Expression
  • Heme / biosynthesis
  • Heme / genetics
  • Humans
  • Hydroxymethylbilane Synthase / genetics*
  • Hydroxymethylbilane Synthase / metabolism
  • Male
  • Mutation, Missense*
  • Porphyria, Acute Intermittent / enzymology
  • Porphyria, Acute Intermittent / genetics*

Substances

  • Heme
  • Hydroxymethylbilane Synthase