Venetoclax plus gilteritinib is effective in preclinical models of <i>FLT3</i> -mutant BCL11B-a lineage-ambiguous leukemia

L Lindsey E. Montefiori (1Department of Pathology, St. Jude Children's Research Hospital, Memphis, TN) I Ilaria Iacobucci (2Department of Pathology, St. Jude Children’s Research Hospital, Memphis, TN) Q Qingsong Gao (2Department of Pathology, St. Jude Children’s Research Hospital, Memphis, TN) J Jamila Moore (2Preclinical Therapeutics Program, St. Jude Children’s Research Hospital, Memphis, TN) W William C. Wright H Huimei Wei (1Department of Pathology, St. Jude Children's Research Hospital, Memphis, TN) P Pradyumna Baviskar (1Department of Pathology, St. Jude Children’s Research Hospital, Memphis, TN) S Surbhi Sona (4Center for Applied Bioinformatics, St. Jude Children’s Research Hospital, Memphis, TN) H Hongjian Jin A Amit Budhraja (5Department of Cell and Molecular Biology, St. Jude Children’s Research Hospital, Memphis, TN) J Josi Lott (6Center of Excellence for Leukemia Studies, St. Jude Children’s Research Hospital, Memphis, TN) Q Qi Zhang Tatarata (7Department of Medicine, State University of New York Downstate Health Sciences University, Brooklyn, NY) Z Zhongshan Cheng T Tanya Khan (1Department of Pathology, St. Jude Children’s Research Hospital, Memphis, TN) E Emily A. Backhaus Wagner (1Department of Pathology, St. Jude Children's Research Hospital, Memphis, TN) M Melissa Johnson (Sarah Cannon Research Institute, Nashville) C Cyrus M. Mehr (1Department of Pathology, St. Jude Children’s Research Hospital, Memphis, TN) B Burgess Freeman L Laura Janke (10Comparative Pathology Core, St. Jude Children’s Research Hospital, Memphis, TN) T Torsten Haferlach (7Munich Leukemia Laboratory, Munich, Germany) P Paul Geeleher P Paul E. Mead (1Department of Pathology, St. Jude Children’s Research Hospital, Memphis, TN) M Marina Konopleva J Joseph T. Opferman (5Department of Cell and Molecular Biology, St. Jude Children’s Research Hospital, Memphis, TN) C Charles G. Mullighan

Abstract

Abstract Aberrant activation of BCL11B (BCL11B-a) defines a subtype of lineage-ambiguous leukemias with T-lymphoid and myeloid features, co-occurring activating FLT3 mutations, and a stem/progenitor immunophenotype and gene expression profile. Similar to other lineage-ambiguous leukemias, optimal treatment is unclear, and there are limited targeted therapeutic options. Here, we investigated the efficacy of B-cell lymphoma 2 (BCL-2) and FMS-like tyrosine kinase 3 (FLT3) inhibition with venetoclax and gilteritinib, respectively, in preclinical models of BCL11B-a leukemia. Despite variation in response to single-agent therapies, the combination of venetoclax plus gilteritinib (VenGilt) was highly effective in all models evaluated. BH3 profiling suggested that resistance to venetoclax monotherapy was due to the tumor-intrinsic dependence on additional BCL-2 family proteins before drug treatment. Longitudinal single-cell RNA sequencing analysis identified mitochondrial pathways and a pro-lymphoid gene expression signature as potential drivers of rare cell survival on VenGilt therapy. These data support clinical evaluation of venetoclax in combination with gilteritinib in BCL11B-a lineage-ambiguous leukemias.

Article Details

Journal Blood
Volume / Issue Vol. 146, Issue 19
Published November 06, 2025
Pages 2350-2356
ISSN 0006-4971
Publisher Elsevier BV

Journal Info

Blood

Elsevier BV

ISSN: 0006-4971 Health Sciences

Authors (25)

L

Lindsey E. Montefiori

1Department of Pathology, St. Jude Children's Research Hospital, Memphis, TN

I

Ilaria Iacobucci

2Department of Pathology, St. Jude Children’s Research Hospital, Memphis, TN

Q

Qingsong Gao

2Department of Pathology, St. Jude Children’s Research Hospital, Memphis, TN

J

Jamila Moore

2Preclinical Therapeutics Program, St. Jude Children’s Research Hospital, Memphis, TN

W

William C. Wright

H

Huimei Wei

1Department of Pathology, St. Jude Children's Research Hospital, Memphis, TN

P

Pradyumna Baviskar

1Department of Pathology, St. Jude Children’s Research Hospital, Memphis, TN

S

Surbhi Sona

4Center for Applied Bioinformatics, St. Jude Children’s Research Hospital, Memphis, TN

H

Hongjian Jin

A

Amit Budhraja

5Department of Cell and Molecular Biology, St. Jude Children’s Research Hospital, Memphis, TN

J

Josi Lott

6Center of Excellence for Leukemia Studies, St. Jude Children’s Research Hospital, Memphis, TN

Q

Qi Zhang Tatarata

7Department of Medicine, State University of New York Downstate Health Sciences University, Brooklyn, NY

Z

Zhongshan Cheng

T

Tanya Khan

1Department of Pathology, St. Jude Children’s Research Hospital, Memphis, TN

E

Emily A. Backhaus Wagner

1Department of Pathology, St. Jude Children's Research Hospital, Memphis, TN

M

Melissa Johnson

Sarah Cannon Research Institute, Nashville

C

Cyrus M. Mehr

1Department of Pathology, St. Jude Children’s Research Hospital, Memphis, TN

B

Burgess Freeman

L

Laura Janke

10Comparative Pathology Core, St. Jude Children’s Research Hospital, Memphis, TN

T

Torsten Haferlach

7Munich Leukemia Laboratory, Munich, Germany

P

Paul Geeleher

P

Paul E. Mead

1Department of Pathology, St. Jude Children’s Research Hospital, Memphis, TN

M

Marina Konopleva

J

Joseph T. Opferman

5Department of Cell and Molecular Biology, St. Jude Children’s Research Hospital, Memphis, TN

C

Charles G. Mullighan