HSCs/MPPs as cells of origin with altered differentiation hierarchy impairing immunomicroenvironment in <i>PML::RARA</i> and <i>CBFα/β</i> fusion AML

N Niu Qiao (Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Centre for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine) Z Zi-Xuan Wang (Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Center for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital Affiliated to Shanghai Jiao Tong University School of Medicine) Y Yu-Liang Zhang (Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Center for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital Affiliated to Shanghai Jiao Tong University School of Medicine) L Liu-Qing-Qing Zhang (Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Center for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital Affiliated to Shanghai Jiao Tong University School of Medicine) H Hong-Ming Zhu (Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Center for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital Affiliated to Shanghai Jiao Tong University School of Medicine) X Xiang-Qin Weng (Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Centre for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine) Y Yong-Mei Zhu (Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Centre for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine) W Wen-Yan Cheng (Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Center for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital Affiliated to Shanghai Jiao Tong University School of Medicine) J Jian-Feng Li (Xiamen University , , ,) L Lu Jiang (School of Flexible Electronics (Future Technologies), Key Laboratory of Flexible Electronics, and Institute of Advanced Materials, Nanjing Tech University, 30 South Puzhu Road, Nanjing 211816, P. R. China) X Xiao-Yu Yan (State Key Laboratory of Quantum Functional Materials, Department of Physics, and Guangdong Basic Research Center of Excellence for Quantum Science, Southern University of Science and Technology) G Guang Yang Y Yang Shen (Beijing National Laboratory for Condensed Matter Physics, Institute of Physics) S Sheng-Yue Wang (Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Centre for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine) Z Zhu Chen (Key Laboratory of Biomedical Polymers of Ministry of Education, Department of Chemistry, Department of Cardiology, Zhongnan Hospital) X Xiao-Jian Sun (Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Center for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital Affiliated to Shanghai Jiao Tong University School of Medicine) F Feng Liu S Sai-Juan Chen (Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Centre for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine)

Abstract

The theoretical possibility for leukemia stem cells (LSCs) to produce both leukemia blasts and dysfunctional immune cells remains underexplored. Here, we investigate three major fusion transcription factor (fTF)-driven acute myeloid leukemia (AML) subtypes [ RUNX1(CBFα)::RUNX1T1 , PML::RARA , and CBFB::MYH11 ] using two optimized single-cell RNA-sequencing technologies to trace fTF expression in 24 de novo AML patients. We demonstrate that the fTFs are widely expressed not only in leukemia blasts but also in differentiated myeloid and lymphoid cells, indicating hematopoietic stem cells or multipotent progenitors (HSCs/MPPs) as LSCs that propagate altered cellular differentiation hierarchies, including immune cells. DNA-FISH confirms the presence of fTFs in T lymphoid and erythroid cells, and targeted sequencing of secondary mutations in sublineages of cells corroborates hierarchical and stepwise leukemogenesis. By tracking RUNX1::RUNX1T1 -expressing cells in patients with or without relapse post–frontline chemotherapy, we highlight the necessity of eradicating LSCs to achieve sustained long-term complete remission and restore a functional immune system capable of suppressing residual disease over time. Comparative single-cell transcriptome analyses further reveal that fTFs are associated with AML subtype-specific differentiation defects in both innate and adaptive immune compartments, suggesting an altered landscape of immune cell–cell communication networks that may facilitate the survival and proliferation of leukemic blasts. Through the examination of intercellular communications among various putative fTF + and normal cell populations, we developed a ligand–receptor (L–R)-based risk-scoring model with independent prognostic value. Collectively, these findings provide insights into the cells of origin of LSCs and the implications of fTF expression for the immune landscape of AML.

Article Details

Volume / Issue Vol. 123, Issue 6
Published February 10, 2026
ISSN 0027-8424
Publisher National Academy of Sciences

Authors (18)

N

Niu Qiao

Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Centre for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine

Z

Zi-Xuan Wang

Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Center for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital Affiliated to Shanghai Jiao Tong University School of Medicine

Y

Yu-Liang Zhang

Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Center for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital Affiliated to Shanghai Jiao Tong University School of Medicine

L

Liu-Qing-Qing Zhang

Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Center for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital Affiliated to Shanghai Jiao Tong University School of Medicine

H

Hong-Ming Zhu

Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Center for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital Affiliated to Shanghai Jiao Tong University School of Medicine

X

Xiang-Qin Weng

Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Centre for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine

Y

Yong-Mei Zhu

Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Centre for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine

W

Wen-Yan Cheng

Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Center for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital Affiliated to Shanghai Jiao Tong University School of Medicine

J

Jian-Feng Li

Xiamen University , , ,

L

Lu Jiang

School of Flexible Electronics (Future Technologies), Key Laboratory of Flexible Electronics, and Institute of Advanced Materials, Nanjing Tech University, 30 South Puzhu Road, Nanjing 211816, P. R. China

X

Xiao-Yu Yan

State Key Laboratory of Quantum Functional Materials, Department of Physics, and Guangdong Basic Research Center of Excellence for Quantum Science, Southern University of Science and Technology

G

Guang Yang

Y

Yang Shen

Beijing National Laboratory for Condensed Matter Physics, Institute of Physics

S

Sheng-Yue Wang

Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Centre for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine

Z

Zhu Chen

Key Laboratory of Biomedical Polymers of Ministry of Education, Department of Chemistry, Department of Cardiology, Zhongnan Hospital

X

Xiao-Jian Sun

Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Center for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital Affiliated to Shanghai Jiao Tong University School of Medicine

F

Feng Liu

S

Sai-Juan Chen

Shanghai Institute of Hematology, State Key Laboratory of Medical Genomics, National Research Centre for Translational Medicine at Shanghai, Research Unit of Hematologic Malignancies Genomics and Translational Research of Chinese Academy of Medical Sciences, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine