POGZ Is Required for Silencing Mouse Embryonic β-like Hemoglobin and Human Fetal Hemoglobin Expression

Cell Rep. 2018 Jun 12;23(11):3236-3248. doi: 10.1016/j.celrep.2018.05.043.

Abstract

Fetal globin genes are transcriptionally silenced during embryogenesis through hemoglobin switching. Strategies to derepress fetal globin expression in the adult could alleviate symptoms in sickle cell disease and β-thalassemia. We identified a zinc-finger protein, pogo transposable element with zinc-finger domain (POGZ), expressed in hematopoietic progenitor cells. Targeted deletion of Pogz in adult hematopoietic cells in vivo results in persistence of embryonic β-like globin expression without affecting erythroid development. POGZ binds to the Bcl11a promoter and erythroid-specific intragenic regulatory regions. Pogz+/- mice show elevated embryonic β-like globin expression, suggesting that partial reduction of Pogz expression results in persistence of embryonic β-like globin expression. Knockdown of POGZ in primary human CD34+ progenitor cell-derived erythroblasts reduces BCL11A expression, a known repressor of embryonic β-like globin expression, and increases fetal hemoglobin expression. These findings are significant, since new therapeutic targets and strategies are needed to treat β-globin disorders.

Keywords: erythropoiesis; fetal globin; gene regulation; globin switching; hematopoietic development; red cells; sickle cell disease; transcription; β-thalassemia.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, N.I.H., Intramural

MeSH terms

  • Animals
  • Carrier Proteins / antagonists & inhibitors
  • Carrier Proteins / genetics
  • Carrier Proteins / metabolism
  • Cell Differentiation
  • Embryo, Mammalian / metabolism
  • Embryonic Development
  • Erythroblasts / cytology
  • Erythroblasts / metabolism
  • Fetal Hemoglobin / genetics
  • Fetal Hemoglobin / metabolism*
  • Hematopoietic Stem Cells / cytology
  • Hematopoietic Stem Cells / metabolism
  • Humans
  • Mice
  • Mice, Inbred C57BL
  • Mice, Knockout
  • Nuclear Proteins / antagonists & inhibitors
  • Nuclear Proteins / genetics
  • Nuclear Proteins / metabolism
  • Promoter Regions, Genetic
  • RNA Interference
  • RNA, Small Interfering / metabolism
  • Repressor Proteins
  • Transposases / antagonists & inhibitors
  • Transposases / genetics*
  • Transposases / metabolism
  • beta-Globins / genetics*
  • beta-Globins / metabolism

Substances

  • BCL11A protein, human
  • Carrier Proteins
  • Nuclear Proteins
  • RNA, Small Interfering
  • Repressor Proteins
  • beta-Globins
  • Fetal Hemoglobin
  • Transposases