Downregulation of ZBTB7A/LRF increases fetal hemoglobin expression in β0-thalassemia/hemoglobin E erythroid cells

Zinc finger and BTB domain-containing 7A (ZBTB7A) is a transcription factor repressor of fetal hemoglobin (HbF; α<sub>2</sub>γ<sub>2</sub>) in erythroid cells. Reactivating γ-globin expression represents a promising therapeutic strategy for β-hemoglobinopathies, including β-thalassemia. While ZBTB7A knockdown is known to elevate HbF levels in HUDEP-2 erythroid cell line and human hematopoietic stem/progenitor cell (HSPC)-derived erythroblasts, its effects in patient-derived cells remain less defined. This study investigates the effects of ZBTB7A downregulation in erythroid cells derived from both β<sup>0</sup> thalassemia/hemoglobin E (β<sup>0</sup>-thal/HbE) patients and healthy donors. ZBTB7A knockdown upregulated embryonic and fetal globin genes (ε-, ζ-, γ-globin), and robust HbF induction while suppressing adult globin gene expression (α-, β-, δ-globin) in both groups. Notably, partial ZBTB7A inhibition was sufficient to achieve HbF reactivation. ZBTB7A depletion delayed erythroid maturation in healthy cells, but not in β⁰-thal/HbE cells, revealing a context-dependent effect on differentiation. These findings support ZBTB7A as a compelling target for β-thalassemia therapy, where partial inhibition could potentially offer therapeutic benefit while minimizing adverse effects on erythroid differentiation.

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Publication Details

Journal
Scientific Reports
Published
2025-12-05
DOI
https://doi.org/10.1038/s41598-025-30762-3
Primary Topic
Hemoglobinopathies and Related Disorders
Type
article
Field-Weighted Citation Impact
0.00

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article

Downregulation of ZBTB7A/LRF increases fetal hemoglobin expression in β0-thalassemia/hemoglobin E erythroid cells

Orapan Sripichai, Sukanya Chumchuen, Chokdee Wongborisuth, Duantida Songdej et al.
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Hemoglobinopathies and Related Disorders
article

Downregulation of ZBTB7A/LRF increases fetal hemoglobin expression in β0-thalassemia/hemoglobin E erythroid cells

Orapan Sripichai, Sukanya Chumchuen, Chokdee Wongborisuth, Duantida Songdej, Suradej Hongeng, Pinyaphat Khamphikham, Amornrat Tangprasittipap, Tanapat Pornsukjantra, Usanarat Anurathapan, Pawarit Innachai
article en

Abstract

Zinc finger and BTB domain-containing 7A (ZBTB7A) is a transcription factor repressor of fetal hemoglobin (HbF; α<sub>2</sub>γ<sub>2</sub>) in erythroid cells. Reactivating γ-globin expression represents a promising therapeutic strategy for β-hemoglobinopathies, including β-thalassemia. While ZBTB7A knockdown is known to elevate HbF levels in HUDEP-2 erythroid cell line and human hematopoietic stem/progenitor cell (HSPC)-derived erythroblasts, its effects in patient-derived cells remain less defined. This study investigates the effects of ZBTB7A downregulation in erythroid cells derived from both β<sup>0</sup> thalassemia/hemoglobin E (β<sup>0</sup>-thal/HbE) patients and healthy donors. ZBTB7A knockdown upregulated embryonic and fetal globin genes (ε-, ζ-, γ-globin), and robust HbF induction while suppressing adult globin gene expression (α-, β-, δ-globin) in both groups. Notably, partial ZBTB7A inhibition was sufficient to achieve HbF reactivation. ZBTB7A depletion delayed erythroid maturation in healthy cells, but not in β⁰-thal/HbE cells, revealing a context-dependent effect on differentiation. These findings support ZBTB7A as a compelling target for β-thalassemia therapy, where partial inhibition could potentially offer therapeutic benefit while minimizing adverse effects on erythroid differentiation.

Scientific ReportsVol. 15(1)
Ministry of Public Health (TH), Mahidol University (TH), Department of Medical Sciences (TH), National Institute of Health of Thailand (TH), Ramathibodi Hospital (TH), Chiang Mai University (TH)
Mahidol University, Ramathibodi Foundation
Openalex Percentile: Top 41%
Hemoglobinopathies and Related Disorders
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