Increased gamma-globin gene expression in beta-thalassemia intermedia patients correlates with a mutation in 3'HS1

Am J Hematol. 2007 Nov;82(11):1005-9. doi: 10.1002/ajh.20979.

Abstract

We report a novel set of genetic markers in the DNaseI hypersensitive sites comprising the human beta-globin locus chromatin hub (CH), namely HS-111 and 3'HS1. The HS-111 (-21 G>A) and 3'HS1 (+179 C>T) transitions form CH haplotypes, which occur at different frequencies in beta-thalassemia intermedia and major patients and normal (nonthalassemic) individuals. We also show that the 3'HS1 (+179 C>T) variation results in a GATA-1 binding site and correlates with increased fetal hemoglobin production in beta-thalassemia intermedia patients. In contrast, the HS-111 (+126 G>A) transition, found in three normal chromosomes, is simply a rare polymorphism. We conclude that the CH haplotypes are useful genetic determinants for beta-thalassemia major and intermedia patients, while the 3'HS1 (+179 C>T) mutation may have functional consequences in gamma-globin genes expression.

Publication types

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

MeSH terms

  • Adult
  • Aged
  • Aged, 80 and over
  • Female
  • Fetal Hemoglobin / genetics
  • Fetal Hemoglobin / metabolism*
  • Gene Expression / genetics
  • Globins / chemistry
  • Globins / genetics*
  • Humans
  • Locus Control Region / genetics*
  • Male
  • Middle Aged
  • Mutation
  • Polymorphism, Single Nucleotide / genetics
  • beta-Thalassemia / genetics*

Substances

  • Globins
  • Fetal Hemoglobin