Genetically Engineered Brain Organoids Recapitulate Spatial and Developmental States of Glioblastoma Progression

Abstract Glioblastoma (GBM) is an aggressive form of brain cancer that is highly resistant to therapy due to significant intra‐tumoral heterogeneity. The lack of robust in vitro models to study early tumor progression has hindered the development of effective therapies. Here, this study develops eng...

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Main Authors: Matthew Ishahak, Rowland H. Han, Devi Annamalai, Timothy Woodiwiss, Colin McCornack, Ryan T. Cleary, Patrick A. DeSouza, Xuan Qu, Sonika Dahiya, Albert H. Kim, Jeffrey R. Millman
Format: Article
Language:English
Published: Wiley 2025-03-01
Series:Advanced Science
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Online Access:https://doi.org/10.1002/advs.202410110
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author Matthew Ishahak
Rowland H. Han
Devi Annamalai
Timothy Woodiwiss
Colin McCornack
Ryan T. Cleary
Patrick A. DeSouza
Xuan Qu
Sonika Dahiya
Albert H. Kim
Jeffrey R. Millman
author_facet Matthew Ishahak
Rowland H. Han
Devi Annamalai
Timothy Woodiwiss
Colin McCornack
Ryan T. Cleary
Patrick A. DeSouza
Xuan Qu
Sonika Dahiya
Albert H. Kim
Jeffrey R. Millman
author_sort Matthew Ishahak
collection DOAJ
description Abstract Glioblastoma (GBM) is an aggressive form of brain cancer that is highly resistant to therapy due to significant intra‐tumoral heterogeneity. The lack of robust in vitro models to study early tumor progression has hindered the development of effective therapies. Here, this study develops engineered GBM organoids (eGBOs) harboring GBM subtype‐specific oncogenic mutations to investigate the underlying transcriptional regulation of tumor progression. Single‐cell and spatial transcriptomic analyses revealed that these mutations disrupt normal neurodevelopment gene regulatory networks resulting in changes in cellular composition and spatial organization. Upon xenotransplantation into immunodeficient mice, eGBOs form tumors that recapitulate the transcriptional and spatial landscape of human GBM samples. Integrative single‐cell trajectory analysis of both eGBO‐derived tumor cells and patient GBM samples reveal the dynamic gene expression changes in developmental cell states underlying tumor progression. This analysis of eGBOs provides an important validation of engineered cancer organoid models and demonstrates their utility as a model of GBM tumorigenesis for future preclinical development of therapeutics.
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spelling doaj-art-4de2491f856d4fb98e7324f098c8fd0b2025-08-20T03:46:46ZengWileyAdvanced Science2198-38442025-03-011210n/an/a10.1002/advs.202410110Genetically Engineered Brain Organoids Recapitulate Spatial and Developmental States of Glioblastoma ProgressionMatthew Ishahak0Rowland H. Han1Devi Annamalai2Timothy Woodiwiss3Colin McCornack4Ryan T. Cleary5Patrick A. DeSouza6Xuan Qu7Sonika Dahiya8Albert H. Kim9Jeffrey R. Millman10Division of Endocrinology Metabolism and Lipid Research Washington University School of Medicine 660 South Euclid Avenue, Campus Box 8127 St. Louis MO 63110 USADepartment of Genetics Washington University School of Medicine 4515 McKinley Ave. St. Louis MO 63110 USADepartment of Genetics Washington University School of Medicine 4515 McKinley Ave. St. Louis MO 63110 USADepartment of Neurological Surgery University of Iowa Healthcare 1800 John Pappajohn Pavilion Iowa City IA 52242 USADepartment of Genetics Washington University School of Medicine 4515 McKinley Ave. St. Louis MO 63110 USADepartment of Genetics Washington University School of Medicine 4515 McKinley Ave. St. Louis MO 63110 USADepartment of Genetics Washington University School of Medicine 4515 McKinley Ave. St. Louis MO 63110 USADepartment of Genetics Washington University School of Medicine 4515 McKinley Ave. St. Louis MO 63110 USADivision of Neuropathology Washington University School of Medicine 660 South Euclid Avenue, Campus Box 8118 St. Louis MO 63110 USADepartment of Genetics Washington University School of Medicine 4515 McKinley Ave. St. Louis MO 63110 USADivision of Endocrinology Metabolism and Lipid Research Washington University School of Medicine 660 South Euclid Avenue, Campus Box 8127 St. Louis MO 63110 USAAbstract Glioblastoma (GBM) is an aggressive form of brain cancer that is highly resistant to therapy due to significant intra‐tumoral heterogeneity. The lack of robust in vitro models to study early tumor progression has hindered the development of effective therapies. Here, this study develops engineered GBM organoids (eGBOs) harboring GBM subtype‐specific oncogenic mutations to investigate the underlying transcriptional regulation of tumor progression. Single‐cell and spatial transcriptomic analyses revealed that these mutations disrupt normal neurodevelopment gene regulatory networks resulting in changes in cellular composition and spatial organization. Upon xenotransplantation into immunodeficient mice, eGBOs form tumors that recapitulate the transcriptional and spatial landscape of human GBM samples. Integrative single‐cell trajectory analysis of both eGBO‐derived tumor cells and patient GBM samples reveal the dynamic gene expression changes in developmental cell states underlying tumor progression. This analysis of eGBOs provides an important validation of engineered cancer organoid models and demonstrates their utility as a model of GBM tumorigenesis for future preclinical development of therapeutics.https://doi.org/10.1002/advs.202410110brain organoidsgenetic engineeringglioblastomasingle‐cell RNA sequencingspatial sequencing
spellingShingle Matthew Ishahak
Rowland H. Han
Devi Annamalai
Timothy Woodiwiss
Colin McCornack
Ryan T. Cleary
Patrick A. DeSouza
Xuan Qu
Sonika Dahiya
Albert H. Kim
Jeffrey R. Millman
Genetically Engineered Brain Organoids Recapitulate Spatial and Developmental States of Glioblastoma Progression
Advanced Science
brain organoids
genetic engineering
glioblastoma
single‐cell RNA sequencing
spatial sequencing
title Genetically Engineered Brain Organoids Recapitulate Spatial and Developmental States of Glioblastoma Progression
title_full Genetically Engineered Brain Organoids Recapitulate Spatial and Developmental States of Glioblastoma Progression
title_fullStr Genetically Engineered Brain Organoids Recapitulate Spatial and Developmental States of Glioblastoma Progression
title_full_unstemmed Genetically Engineered Brain Organoids Recapitulate Spatial and Developmental States of Glioblastoma Progression
title_short Genetically Engineered Brain Organoids Recapitulate Spatial and Developmental States of Glioblastoma Progression
title_sort genetically engineered brain organoids recapitulate spatial and developmental states of glioblastoma progression
topic brain organoids
genetic engineering
glioblastoma
single‐cell RNA sequencing
spatial sequencing
url https://doi.org/10.1002/advs.202410110
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