Preclinical study of microphthalmia-associated transcription factor inhibitor ML329 in gastrointestinal stromal tumor growth

Gastrointestinal stromal tumors (GISTs) comprise about 80% of mesenchymal neoplasms in the gastrointestinal tract. Although imatinib mesylate is the preferred treatment, the development of drug resistance highlights the need for novel therapeutic strategies. Recently, we have identified the micropht...

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Main Authors: Mario Guerrero, Elizabeth Proaño-Pérez, Eva Serrano-Candelas, Alfonso García-Valverde, Berenice Carrillo-Rodríguez, Jordi Rosell, César Serrano, Margarita Martin
Format: Article
Language:English
Published: Elsevier 2025-06-01
Series:Molecular Therapy: Oncology
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Online Access:http://www.sciencedirect.com/science/article/pii/S2950329925000529
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author Mario Guerrero
Elizabeth Proaño-Pérez
Eva Serrano-Candelas
Alfonso García-Valverde
Berenice Carrillo-Rodríguez
Jordi Rosell
César Serrano
Margarita Martin
author_facet Mario Guerrero
Elizabeth Proaño-Pérez
Eva Serrano-Candelas
Alfonso García-Valverde
Berenice Carrillo-Rodríguez
Jordi Rosell
César Serrano
Margarita Martin
author_sort Mario Guerrero
collection DOAJ
description Gastrointestinal stromal tumors (GISTs) comprise about 80% of mesenchymal neoplasms in the gastrointestinal tract. Although imatinib mesylate is the preferred treatment, the development of drug resistance highlights the need for novel therapeutic strategies. Recently, we have identified the microphthalmia-associated transcription factor (MITF) as a critical player in pro-survival signaling and tumor growth. This study investigates the effects of MITF inhibition using ML329, an MITF pathway inhibitor, on GIST cell viability in vitro and in NMRI-nu/nu mouse xenograft models. ML329 suppresses growth in imatinib-sensitive (GIST-T1) and -resistant (GIST 430/654) cell lines, impairs MITF targets such as BCL2 and CDK2, and induces S-G2/M cell-cycle arrest. In vivo, ML329 is well tolerated and significantly reduces tumor growth in established imatinib-sensitive and -resistant GIST models. These findings underscore the importance of MITF in GIST growth and support its inhibition as a promising therapeutic approach.
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series Molecular Therapy: Oncology
spelling doaj-art-b7e593a563254c3b9f340a628fee1ade2025-08-20T02:18:29ZengElsevierMolecular Therapy: Oncology2950-32992025-06-0133220098310.1016/j.omton.2025.200983Preclinical study of microphthalmia-associated transcription factor inhibitor ML329 in gastrointestinal stromal tumor growthMario Guerrero0Elizabeth Proaño-Pérez1Eva Serrano-Candelas2Alfonso García-Valverde3Berenice Carrillo-Rodríguez4Jordi Rosell5César Serrano6Margarita Martin7Biochemistry and Molecular Biology Unit, Biomedicine Department, Faculty of Medicine and Health Sciences, University of Barcelona, 08036 Barcelona, SpainBiochemistry and Molecular Biology Unit, Biomedicine Department, Faculty of Medicine and Health Sciences, University of Barcelona, 08036 Barcelona, Spain; Multidisciplinary and Translational Research in Inflammation and Immunoallergy (METRI2 A), Institut d'Investigacions Biomediques August Pi i Sunyer (IDIBAPS), 08036 Barcelona, Spain; Facultad de Ciencias de la Salud, Universidad Técnica de Ambato, Ambato 180105, Ecuador; Nutrigenx, Universidad Técnica de Ambato, Ambato 180105, EcuadorBiochemistry and Molecular Biology Unit, Biomedicine Department, Faculty of Medicine and Health Sciences, University of Barcelona, 08036 Barcelona, Spain; Multidisciplinary and Translational Research in Inflammation and Immunoallergy (METRI2 A), Institut d'Investigacions Biomediques August Pi i Sunyer (IDIBAPS), 08036 Barcelona, SpainSarcoma Translational Research Program, Vall d’Hebron Institute of Oncology (VHIO), Vall d’Hebron University Hospital, 08035 Barcelona, SpainBiochemistry and Molecular Biology Unit, Biomedicine Department, Faculty of Medicine and Health Sciences, University of Barcelona, 08036 Barcelona, SpainSarcoma Translational Research Program, Vall d’Hebron Institute of Oncology (VHIO), Vall d’Hebron University Hospital, 08035 Barcelona, SpainSarcoma Translational Research Program, Vall d’Hebron Institute of Oncology (VHIO), Vall d’Hebron University Hospital, 08035 Barcelona, Spain; Department of Medical Oncology, Vall d'Hebron University Hospital, 08035 Barcelona, SpainBiochemistry and Molecular Biology Unit, Biomedicine Department, Faculty of Medicine and Health Sciences, University of Barcelona, 08036 Barcelona, Spain; Multidisciplinary and Translational Research in Inflammation and Immunoallergy (METRI2 A), Institut d'Investigacions Biomediques August Pi i Sunyer (IDIBAPS), 08036 Barcelona, Spain; Corresponding author: Margarita Martin, Biochemistry and Molecular Biology Unit, Biomedicine Department, Faculty of Medicine and Health Sciences, University of Barcelona, 08036 Barcelona, Spain.Gastrointestinal stromal tumors (GISTs) comprise about 80% of mesenchymal neoplasms in the gastrointestinal tract. Although imatinib mesylate is the preferred treatment, the development of drug resistance highlights the need for novel therapeutic strategies. Recently, we have identified the microphthalmia-associated transcription factor (MITF) as a critical player in pro-survival signaling and tumor growth. This study investigates the effects of MITF inhibition using ML329, an MITF pathway inhibitor, on GIST cell viability in vitro and in NMRI-nu/nu mouse xenograft models. ML329 suppresses growth in imatinib-sensitive (GIST-T1) and -resistant (GIST 430/654) cell lines, impairs MITF targets such as BCL2 and CDK2, and induces S-G2/M cell-cycle arrest. In vivo, ML329 is well tolerated and significantly reduces tumor growth in established imatinib-sensitive and -resistant GIST models. These findings underscore the importance of MITF in GIST growth and support its inhibition as a promising therapeutic approach.http://www.sciencedirect.com/science/article/pii/S2950329925000529MT: Regular IssueMITFcell survivalcell cyclegastrointestinal stromal tumors
spellingShingle Mario Guerrero
Elizabeth Proaño-Pérez
Eva Serrano-Candelas
Alfonso García-Valverde
Berenice Carrillo-Rodríguez
Jordi Rosell
César Serrano
Margarita Martin
Preclinical study of microphthalmia-associated transcription factor inhibitor ML329 in gastrointestinal stromal tumor growth
Molecular Therapy: Oncology
MT: Regular Issue
MITF
cell survival
cell cycle
gastrointestinal stromal tumors
title Preclinical study of microphthalmia-associated transcription factor inhibitor ML329 in gastrointestinal stromal tumor growth
title_full Preclinical study of microphthalmia-associated transcription factor inhibitor ML329 in gastrointestinal stromal tumor growth
title_fullStr Preclinical study of microphthalmia-associated transcription factor inhibitor ML329 in gastrointestinal stromal tumor growth
title_full_unstemmed Preclinical study of microphthalmia-associated transcription factor inhibitor ML329 in gastrointestinal stromal tumor growth
title_short Preclinical study of microphthalmia-associated transcription factor inhibitor ML329 in gastrointestinal stromal tumor growth
title_sort preclinical study of microphthalmia associated transcription factor inhibitor ml329 in gastrointestinal stromal tumor growth
topic MT: Regular Issue
MITF
cell survival
cell cycle
gastrointestinal stromal tumors
url http://www.sciencedirect.com/science/article/pii/S2950329925000529
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