HDAC7 induction combined with standard-of-care chemotherapy provides a therapeutic advantage in t(4;11) infant B-cell acute lymphoblastic leukemia

Abstract Background Infants diagnosed with B cell acute lymphoblastic leukemia (B-ALL) and t(4;11) chromosomal rearrangement display poor therapeutic response, associated to the low expression of B lymphocyte factor HDAC7. This study was conceived to identify a therapeutic strategy for t(4;11) B-ALL...

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Main Authors: Oriol de Barrios, Ingrid Ocón-Gabarró, Mar Gusi-Vives, Olga Collazo, Ainara Meler, Paola A. Romecín, Alba Martínez-Moreno, Juan Ramón Tejedor, Mario F. Fraga, Pauline Schneider, Michela Bardini, Giovanni Cazzaniga, Rolf Marschalek, Ronald W. Stam, Clara Bueno, Pablo Menéndez, Maribel Parra
Format: Article
Language:English
Published: BMC 2025-07-01
Series:Biomarker Research
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Online Access:https://doi.org/10.1186/s40364-025-00810-1
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Summary:Abstract Background Infants diagnosed with B cell acute lymphoblastic leukemia (B-ALL) and t(4;11) chromosomal rearrangement display poor therapeutic response, associated to the low expression of B lymphocyte factor HDAC7. This study was conceived to identify a therapeutic strategy for t(4;11) B-ALL that restores optimal HDAC7 expression. Methods A multiomics approach in a large infant pro-B-ALL cohort was employed to identify HDAC7’s repression mechanism. These data, combined with cell culture assays in a variety of pro-B-ALL cell lines with differential HDAC7 levels, led us to define a novel combination therapy. Murine leukemia models and ex vivo assays using patient-derived xenografts (PDX) were employed to assess the benefits of this therapy when incorporated to glucocorticoid-based chemotherapy. Results Our data demonstrates that HDAC7 is epigenetically silenced by EZH2 and KMT2A::AFF1 fusion protein. Remarkably, the Menin-1 inhibitor MI-538 restores HDAC7 expression, and the effect is enhanced by class I HDAC inhibitor chidamide. This treatment drives leukemic pro-B cells towards a more differentiated state and impairs aberrant proliferation in an HDAC7-dependent manner. This newly identified therapy increases glucocorticoid sensitivity of PDX cells ex vivo, by repressing RUNX2 transcription factor. Finally, combining MI-538 and chidamide with standard chemotherapy reduces PDX cells engraftment in vivo and delays relapse. Conclusions The combined therapy proposed, based on Menin-1 inhibition, improves t(4;11) B-ALL cells’ response to standard therapy, an effect partially mediated by HDAC7 induction. Therefore, this novel therapy opens a new field for personalized treatments in high-risk leukemia, especially for infants presenting low expression of HDAC7 B cell factor.
ISSN:2050-7771