Increased gamma-globin expression in a nondeletion HPFH mediated by an erythroid-specific DNA-binding factor

Nature. 1989 Mar 30;338(6214):435-8. doi: 10.1038/338435a0.

Abstract

In man, a shift from gamma- to beta-globin gene expression in erythroblasts underlies a switch from fetal to adult haemoglobin during development. In hereditary persistence of fetal haemoglobin (HPFH), inappropriately high gamma-globin expression in adult life is associated with deletions in the beta-globin cluster or with single-base changes upstream of the gamma-globin genes. To account for enhanced gamma-gene expression in HPFH of the non-deletion type, we tested the nuclear proteins of human erythroleukaemia cells that bind gamma-promoter sequences in vitro by correlating specific mutations in their binding sites with promoter activity. An erythroid-specific factor (GF-1) binds as a single molecule to the -195 to -170 region and contacts two TATCT(AGATA) motifs, but not the conserved octamer (ATGCAAAT) that separates them. We observe that a single change (at -175, T----C) found in HPFH leads to increased promoter activity only in erythroid cells. This effect is mediated by GF-1, the human counterpart of the chicken erythroid factor Eryf 1. The form of HPFH we studied here is an inherited disorder which can be ascribed to the action of a cell-specific DNA-binding factor on a mutant promoter.

Publication types

  • Research Support, Non-U.S. Gov't
  • Research Support, U.S. Gov't, P.H.S.

MeSH terms

  • DNA-Binding Proteins / genetics*
  • Erythroid-Specific DNA-Binding Factors
  • Fetal Hemoglobin / genetics*
  • Globins / genetics*
  • Hemoglobinopathies / genetics*
  • Humans
  • Leukemia, Erythroblastic, Acute / genetics
  • Promoter Regions, Genetic
  • Transcription Factors*

Substances

  • DNA-Binding Proteins
  • Erythroid-Specific DNA-Binding Factors
  • Transcription Factors
  • Globins
  • Fetal Hemoglobin