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BCL11A-deficient human erythropoiesis is impaired in vitro and after xenotransplantation into mice

Yoonjeong Jang, Ruopeng Feng, Lance E. Palmer, Thiyagaraj Mayuranathan, Yu Yao, Kalin Mayberry, Sheng Zhou, Jian Xu, Jeffrey M. Gossett, Guolian Kang, Yong Cheng, Jonathan Yen, Mitchell J. Weiss

2025Blood Advances15 citationsDOIOpen Access PDF

Abstract

ABSTRACT: Genetic depletion of the transcriptional repressor BCL11A in red blood cell precursors alleviates β-hemoglobinopathies by inducing the fetal γ-globin genes. However, additional erythroid genes are regulated by BCL11A and the effects of its deficiency on erythropoiesis are insufficiently described. We discovered that Cas9 disruption of the BCL11A intron 2 erythroid enhancer in CD34+ hematopoietic stem and progenitor cells using a clinically approved strategy caused impaired expansion and apoptosis of erythroid precursors in vitro and reduced repopulation of the erythroid compartment after xenotransplantation into immunodeficient mice. Mutant colony-forming unit erythroid cells, proerythroblasts, and basophilic erythroblasts exhibited dysregulation of 94 genes (more than twofold change, false discovery rate < 0.05), 25 of which are likely direct targets of BCL11A. Differentially expressed genes were associated with a range of biological pathways that affect cell expansion and survival. Our findings reveal that BCL11A regulates additional aspects of erythropoiesis beyond γ-globin gene repression, with unknown clinical consequences.

Topics & Concepts

ErythropoiesisBiologyHaematopoiesisXenotransplantationEnhancerCell biologyCancer researchProgenitor cellStem cellGeneMolecular biologyGeneticsGene expressionTransplantationInternal medicineAnemiaMedicineHemoglobinopathies and Related DisordersErythrocyte Function and PathophysiologyEpigenetics and DNA Methylation
BCL11A-deficient human erythropoiesis is impaired in vitro and after xenotransplantation into mice | Litcius