Cell Reports · 2018 · 39 citations · 43 references
Life SciencesGeneticsMolecular GeneticsDevelopmental Cell BiologyEmbryogenesisGene ExpressionHematopoietic DevelopmentHematopoietic DifferentiationHematopoietic Stem Cell BiologyRed Blood Cell DisordersRed Blood Cell PhysiologySickle Cell DiseaseHemoglobin SwitchingProgenitor CellsSickle Cell AnemiaZinc Binding ProteinsZinc Finger DomainsZinc Finger MotifsZinc Finger ProteinsZinc Finger Transcription Factors
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.
43
Inducible Gene Targeting in Mice
Ralf Kühn, Frieder Schwenk, Michel Aguet et al. · Science · 1995 · 1.9K citations · Full text
De Novo Gene Disruptions in Children on the Autistic Spectrum
Ivan Iossifov, Michael Ronemus, Dan Levy et al. · Neuron · 2012 · 1.5K citations · Full text