Transposable elements as novel therapeutic targets for PARPi-induced synthetic lethality in PcG-mutated blood cancer

B Bernd B. Zeisig (1Comprehensive Cancer Centre, King’s College London, London, United Kingdom) C Chiou-Tsun Tsai C Clemence Virely (1Comprehensive Cancer Centre, King’s College London, London, United Kingdom) T Tsz Kan Fung (1King's College London, Comprehensive Cancer Center, London, United Kingdom) A Ali Tuğrul Akin (1Comprehensive Cancer Centre, King’s College London, London, United Kingdom) E Estelle Troadec (1Comprehensive Cancer Centre, King’s College London, London, United Kingdom) B Bo Jiao (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) I I. Richard Thompson P Priscilla Nga Ieng Lau (1Comprehensive Cancer Centre, King’s College London, London, United Kingdom) N Nanjun Li (1Comprehensive Cancer Centre, King’s College London, London, United Kingdom) A Andriani Charalambous (1Comprehensive Cancer Centre, King’s College London, London, United Kingdom) L Larissa Bomfim (1King's College London, Comprehensive Cancer Center, London, United Kingdom) J Jennifer Lynch (Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, United States) A Athina Georgiou (1Comprehensive Cancer Centre, King’s College London, London, United Kingdom) R Robbert Hoogeboom (1Comprehensive Cancer Centre, King’s College London, London, United Kingdom) C Claire Lynn (1Comprehensive Cancer Centre, King’s College London, London, United Kingdom) S Si-Yi Zhang (Hunan Provincial Key Laboratory of Flexible Electronic Materials Genome Engineering, School of Physics and Electronic Science, Changsha University of Science and Technology 1 , Changsha 410114,) P Piers E. M. Patten (7Department of Haematology, Comprehensive Cancer Centre, King’s College London, London, United Kingdom) C Cynthia L. Fisher (5School of Biomedical Engineering, The University of British Columbia, Vancouver, Canada) A Anna Schuh S Seishi Ogawa G Ghulam Mufti (43King's College, London, United Kingdom) M Mohammad M. Karimi C Chi Wai Eric So (1Comprehensive Cancer Centre, King’s College London, London, United Kingdom)

Abstract

Abstract Loss-of-function (LoF) mutations frequently found in human cancers are generally intractable by classical small molecule inhibitor approaches. Among them are mutations affecting Polycomb-group (PcG) epigenetic regulators, enhancer of zeste homolog 2 (EZH2) and Additional sex combs like 1 (ASXL1), frequently found in hematological malignancies of myeloid or lymphoid lineage, and their concurrent mutations associates with particularly poor prognosis. Although there is a clear need to develop novel and effective treatments for these patients, the lack of appropriate disease models and mechanistic insights have significantly hindered the progress. Here, we show that genetic inactivation of Asxl1 and Ezh2 in murine hematopoietic stem/progenitor cells results in highly penetrant hematological malignancies as observed in corresponding human diseases. These PcG proteins regulate both coding and noncoding genomes, leading to marked reactivation of transposable elements (TEs) and DNA damage responses in PcG LoF–mutated cells, which create a novel vulnerability for poly(ADP-ribose) polymerase (PARP) inhibitor (PARPi)–induced synthetic lethality. Using both mouse models and primary patient samples, we demonstrate that Asxl1/Ezh2-mutated cells are highly sensitive to PARPis that induce excessive DNA damage and significantly extend disease latency. Intriguingly, the observed PARPi sensitivity can be specifically overridden by reverse transcriptase inhibitors that interrupt target site–primed reverse transcription and life cycle of TEs. This mechanism is contrastingly different from the current concept of BRCAness associated PARPi-induced synthetic lethality, which largely rely on deficient homologous recombination, and is independent on reverse transcriptase inhibitors. Together, this study reveals a novel application and mechanism of PARPi-induced synthetic lethal targeting of blood cancers with reactivated TEs such as those carrying PcG epigenetic mutations.

Article Details

Journal Blood
Volume / Issue Vol. 146, Issue 11
Published September 11, 2025
Pages 1314-1330
ISSN 0006-4971
Publisher Elsevier BV

Journal Info

Blood

Elsevier BV

ISSN: 0006-4971 Health Sciences

Authors (24)

B

Bernd B. Zeisig

1Comprehensive Cancer Centre, King’s College London, London, United Kingdom

C

Chiou-Tsun Tsai

C

Clemence Virely

1Comprehensive Cancer Centre, King’s College London, London, United Kingdom

T

Tsz Kan Fung

1King's College London, Comprehensive Cancer Center, London, United Kingdom

A

Ali Tuğrul Akin

1Comprehensive Cancer Centre, King’s College London, London, United Kingdom

E

Estelle Troadec

1Comprehensive Cancer Centre, King’s College London, London, United Kingdom

B

Bo Jiao

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

I

I. Richard Thompson

P

Priscilla Nga Ieng Lau

1Comprehensive Cancer Centre, King’s College London, London, United Kingdom

N

Nanjun Li

1Comprehensive Cancer Centre, King’s College London, London, United Kingdom

A

Andriani Charalambous

1Comprehensive Cancer Centre, King’s College London, London, United Kingdom

L

Larissa Bomfim

1King's College London, Comprehensive Cancer Center, London, United Kingdom

J

Jennifer Lynch

Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, United States

A

Athina Georgiou

1Comprehensive Cancer Centre, King’s College London, London, United Kingdom

R

Robbert Hoogeboom

1Comprehensive Cancer Centre, King’s College London, London, United Kingdom

C

Claire Lynn

1Comprehensive Cancer Centre, King’s College London, London, United Kingdom

S

Si-Yi Zhang

Hunan Provincial Key Laboratory of Flexible Electronic Materials Genome Engineering, School of Physics and Electronic Science, Changsha University of Science and Technology 1 , Changsha 410114,

P

Piers E. M. Patten

7Department of Haematology, Comprehensive Cancer Centre, King’s College London, London, United Kingdom

C

Cynthia L. Fisher

5School of Biomedical Engineering, The University of British Columbia, Vancouver, Canada

A

Anna Schuh

S

Seishi Ogawa

G

Ghulam Mufti

43King's College, London, United Kingdom

M

Mohammad M. Karimi

C

Chi Wai Eric So

1Comprehensive Cancer Centre, King’s College London, London, United Kingdom