Identification of a small molecule targeting EPLIN as a novel strategy for the treatment of pediatric neuroblastoma and medulloblastoma

Abstract Amplification of the MYCN proto-oncogene serves as a key marker of aggressive disease and poor treatment outcomes in certain pediatric tumors originating from the nervous system, including neuroblastoma and medulloblastoma. However, the complex nature of the challenging MYCN protein undersc...

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Main Authors: Emma Lindell, Jing Guo, Miao Zhao, Natallia Rameika, Xi Lu, Tabea Wacker, Lei Zhong, Tobias Bergström, Sara Svanberg, Azazul I. Chowdhury, Peter Bergsten, Xingqi Chen, Daniel Bexell, Fredrik J. Swartling, Tobias Sjöblom, Xiaonan Zhang
Format: Article
Language:English
Published: Nature Publishing Group 2025-07-01
Series:Cell Death and Disease
Online Access:https://doi.org/10.1038/s41419-025-07876-7
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author Emma Lindell
Jing Guo
Miao Zhao
Natallia Rameika
Xi Lu
Tabea Wacker
Lei Zhong
Tobias Bergström
Sara Svanberg
Azazul I. Chowdhury
Peter Bergsten
Xingqi Chen
Daniel Bexell
Fredrik J. Swartling
Tobias Sjöblom
Xiaonan Zhang
author_facet Emma Lindell
Jing Guo
Miao Zhao
Natallia Rameika
Xi Lu
Tabea Wacker
Lei Zhong
Tobias Bergström
Sara Svanberg
Azazul I. Chowdhury
Peter Bergsten
Xingqi Chen
Daniel Bexell
Fredrik J. Swartling
Tobias Sjöblom
Xiaonan Zhang
author_sort Emma Lindell
collection DOAJ
description Abstract Amplification of the MYCN proto-oncogene serves as a key marker of aggressive disease and poor treatment outcomes in certain pediatric tumors originating from the nervous system, including neuroblastoma and medulloblastoma. However, the complex nature of the challenging MYCN protein underscores the urgent need for additional targets and therapies to tackle neuroblastoma and medulloblastoma. In this study, with a primary focus on neuroblastoma and the aim of also benefiting children with medulloblastoma, we identified FLIX5, a small compound that exhibits broad cytotoxicity against both neuroblastoma and medulloblastoma cells, primarily by triggering apoptosis. Furthermore, FLIX5 enhances the cholesterol dependency of neuroblastoma cells under conditions where mitochondrial function is impaired. FLIX5 as well shows a synergistic effect when combined with vincristine, a conventional anticancer drug, against neuroblastoma cells and organoids. Through proteome integral solubility alteration, computational molecular docking predictions, and cellular thermal shift assays for target identification and validation, FLIX5 reveals EPLIN (Epithelial Protein Lost In Neoplasm) as a previously unexplored drug target. EPLIN is involved in several cellular processes, including cholesterol uptake and mitochondrial function. The discovery of FLIX5 targeting EPLIN presents new opportunities for treating malignant pediatric tumors, with the potential to target chemoresistant dormant cancer cells and broaden its therapeutic applications to other tumor types.
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spelling doaj-art-567c6c3fbde5471ebd08c0425c566a792025-08-20T03:46:20ZengNature Publishing GroupCell Death and Disease2041-48892025-07-0116111510.1038/s41419-025-07876-7Identification of a small molecule targeting EPLIN as a novel strategy for the treatment of pediatric neuroblastoma and medulloblastomaEmma Lindell0Jing Guo1Miao Zhao2Natallia Rameika3Xi Lu4Tabea Wacker5Lei Zhong6Tobias Bergström7Sara Svanberg8Azazul I. Chowdhury9Peter Bergsten10Xingqi Chen11Daniel Bexell12Fredrik J. Swartling13Tobias Sjöblom14Xiaonan Zhang15Department of Immunology, Genetics and Pathology, Uppsala UniversityCentre for Computational Biology, Duke-NUS Medical SchoolDepartment of Immunology, Genetics and Pathology, Uppsala UniversityDepartment of Immunology, Genetics and Pathology, Uppsala UniversityDepartment of Immunology, Genetics and Pathology, Uppsala UniversityDepartment of Immunology, Genetics and Pathology, Uppsala UniversityDepartment of Immunology, Genetics and Pathology, Uppsala UniversityDepartment of Immunology, Genetics and Pathology, Uppsala UniversityDepartment of Immunology, Genetics and Pathology, Uppsala UniversityDepartment of Medical Cell Biology, Uppsala University, BMC, Husargatan 3Department of Medical Cell Biology, Uppsala University, BMC, Husargatan 3Department of Immunology, Genetics and Pathology, Uppsala UniversityDivision of Translational Cancer Research, Department of Laboratory Medicine, Lund UniversityDepartment of Immunology, Genetics and Pathology, Uppsala UniversityDepartment of Immunology, Genetics and Pathology, Uppsala UniversityDepartment of Immunology, Genetics and Pathology, Uppsala UniversityAbstract Amplification of the MYCN proto-oncogene serves as a key marker of aggressive disease and poor treatment outcomes in certain pediatric tumors originating from the nervous system, including neuroblastoma and medulloblastoma. However, the complex nature of the challenging MYCN protein underscores the urgent need for additional targets and therapies to tackle neuroblastoma and medulloblastoma. In this study, with a primary focus on neuroblastoma and the aim of also benefiting children with medulloblastoma, we identified FLIX5, a small compound that exhibits broad cytotoxicity against both neuroblastoma and medulloblastoma cells, primarily by triggering apoptosis. Furthermore, FLIX5 enhances the cholesterol dependency of neuroblastoma cells under conditions where mitochondrial function is impaired. FLIX5 as well shows a synergistic effect when combined with vincristine, a conventional anticancer drug, against neuroblastoma cells and organoids. Through proteome integral solubility alteration, computational molecular docking predictions, and cellular thermal shift assays for target identification and validation, FLIX5 reveals EPLIN (Epithelial Protein Lost In Neoplasm) as a previously unexplored drug target. EPLIN is involved in several cellular processes, including cholesterol uptake and mitochondrial function. The discovery of FLIX5 targeting EPLIN presents new opportunities for treating malignant pediatric tumors, with the potential to target chemoresistant dormant cancer cells and broaden its therapeutic applications to other tumor types.https://doi.org/10.1038/s41419-025-07876-7
spellingShingle Emma Lindell
Jing Guo
Miao Zhao
Natallia Rameika
Xi Lu
Tabea Wacker
Lei Zhong
Tobias Bergström
Sara Svanberg
Azazul I. Chowdhury
Peter Bergsten
Xingqi Chen
Daniel Bexell
Fredrik J. Swartling
Tobias Sjöblom
Xiaonan Zhang
Identification of a small molecule targeting EPLIN as a novel strategy for the treatment of pediatric neuroblastoma and medulloblastoma
Cell Death and Disease
title Identification of a small molecule targeting EPLIN as a novel strategy for the treatment of pediatric neuroblastoma and medulloblastoma
title_full Identification of a small molecule targeting EPLIN as a novel strategy for the treatment of pediatric neuroblastoma and medulloblastoma
title_fullStr Identification of a small molecule targeting EPLIN as a novel strategy for the treatment of pediatric neuroblastoma and medulloblastoma
title_full_unstemmed Identification of a small molecule targeting EPLIN as a novel strategy for the treatment of pediatric neuroblastoma and medulloblastoma
title_short Identification of a small molecule targeting EPLIN as a novel strategy for the treatment of pediatric neuroblastoma and medulloblastoma
title_sort identification of a small molecule targeting eplin as a novel strategy for the treatment of pediatric neuroblastoma and medulloblastoma
url https://doi.org/10.1038/s41419-025-07876-7
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