ArticleCell stem cell2019
Engineering Genetic Predisposition in Human Neuroepithelial Stem Cells Recapitulates Medulloblastoma Tumorigenesis.
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Article in Cell stem cell, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 58 papers.
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58citing papers in PubMed
3.8field-weighted citation impact, top 6% of its field
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Who cites it
58 citing papers in PubMed, 84 citations in OpenAlex.
- Modelling brain tumours with organoids: towards precision medicine in neuro-oncology.Nature reviews. Neurology · 2026Review
- Medulloblastoma stem cell programs: Molecular roadmaps of disease progression.Developmental cell · 2026Review
- Modeling pediatric brain tumors with human stem cells.Frontiers in cellular neuroscience · 2026Review
- Human stem cell models for group 3 medulloblastoma uncover JARID1B as a regulator of the chromatin landscape.bioRxiv : the preprint server for biology · 2025Article
- Epigenomic landscape of the developing human rhombic lip reveals gene regulatory network and non-coding loci of developmental, evolutionary, and disease relevance.bioRxiv : the preprint server for biology · 2025Article
- Current State-of-the-Art Animal Models of Pediatric Brain Tumors.Brain sciences · 2025Review
- Single-cell transcriptomic sequencing identifies subcutaneous patient-derived xenograft recapitulated medulloblastoma.Animal models and experimental medicine · 2025Article
- Arrested development: the dysfunctional life history of medulloblastoma.Genes & development · 2025Review
- Embryonic Stem Cell Models of Human Brain Tumors.Methods in molecular biology (Clifton, N.J.) · 2025Article
- Brain tumors and induced pluripotent stem cell technology: a systematic review of the literature.Annals of medicine and surgery (2012) · 2025Article
- Tryptamine-Functionalized Lipid Nanocarriers Co-delivering SMO/BRD4 Inhibitors for Synergistic Medulloblastoma Therapy.Biomaterials research · 2025Article
- Human stem cell models to unravel brain cancer.BMC cancer · 2024Review
- Comparison of human pluripotent stem cell differentiation protocols to generate neuroblastoma tumors.Scientific reports · 2024Article
- GRP78 inhibitor YUM70 upregulates 4E-BP1 and suppresses c-MYC expression and viability of oncogenic c-MYC tumors.Neoplasia (New York, N.Y.) · 2024Article
- PRDM6 promotes medulloblastoma by repressing chromatin accessibility and altering gene expression.Scientific reports · 2024Article
- A human neural crest model reveals the developmental impact of neuroblastoma-associated chromosomal aberrations.Nature communications · 2024Article
- LOXL1-AS1 contributes to metastasis in sonic-hedgehog medulloblastoma by promoting cancer stem-like phenotypes.Journal of experimental & clinical cancer research : CR · 2024Article
- ALK upregulates POSTN and WNT signaling to drive neuroblastoma.Cell reports · 2024Article
- PTCH1-mutant human cerebellar organoids exhibit altered neural development and recapitulate early medulloblastoma tumorigenesis.Disease models & mechanisms · 2024Article
- GSE1 links the HDAC1/CoREST co-repressor complex to DNA damage.Nucleic acids research · 2023Article
Corrections and comments
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Authors and funding
38 authors at 18 institutions in 6 countries.
Miller HuangDepartment of Neurology and the Helen Diller Family Comprehensive Cancer Center, University of California, San Francisco, San Francisco, CA 94158, USA.
Jignesh TailorWellcome Trust-MRC Stem Cell Institute, University of Cambridge, Tennis Court Road, Cambridge CB2 1QR, UK; Institute of Cancer Research, Sutton, London SM2 5NG, UK; Developmental & Stem Cell Biology Program, The Hospital for Sick Children, Toronto, ON, Canada; Division of Neurosurgery, The Hospital for Sick Children, Toronto, ON, Canada.
Qiqi ZhenDepartment of Neurology and the Helen Diller Family Comprehensive Cancer Center, University of California, San Francisco, San Francisco, CA 94158, USA.
Aaron H GillmorDepartment of Biochemistry and Molecular Biology, University of Calgary, Calgary, AB, Canada; Charbonneau Cancer Institute, University of Calgary, Calgary, AB, Canada; Alberta Children's Hospital Research Institute, Calgary, AB, Canada.
Matthew L MillerDepartment of Neurology and the Helen Diller Family Comprehensive Cancer Center, University of California, San Francisco, San Francisco, CA 94158, USA.
Holger WeishauptDepartment of Immunology, Genetics and Pathology, Science for Life Laboratory, Uppsala University, 751 85 Uppsala, Sweden.
Justin ChenDepartment of Genetics, Stanford University School of Medicine, Stanford, CA 94305, USA.
Tina ZhengDepartment of Neurology and the Helen Diller Family Comprehensive Cancer Center, University of California, San Francisco, San Francisco, CA 94158, USA.
Emily K NashDepartment of Neurology and the Helen Diller Family Comprehensive Cancer Center, University of California, San Francisco, San Francisco, CA 94158, USA.
Lauren K McHenryDepartment of Neurology and the Helen Diller Family Comprehensive Cancer Center, University of California, San Francisco, San Francisco, CA 94158, USA.
Zhenyi AnDepartment of Neurology and the Helen Diller Family Comprehensive Cancer Center, University of California, San Francisco, San Francisco, CA 94158, USA.
Fubaiyang YeDepartment of Neurology and the Helen Diller Family Comprehensive Cancer Center, University of California, San Francisco, San Francisco, CA 94158, USA.
Yasuhiro TakashimaWellcome Trust-MRC Stem Cell Institute, University of Cambridge, Tennis Court Road, Cambridge CB2 1QR, UK.
James ClarkeWellcome Trust-MRC Stem Cell Institute, University of Cambridge, Tennis Court Road, Cambridge CB2 1QR, UK.
Harold AyeteyWellcome Trust-MRC Stem Cell Institute, University of Cambridge, Tennis Court Road, Cambridge CB2 1QR, UK.
Florence M G CavalliDevelopmental & Stem Cell Biology Program, The Hospital for Sick Children, Toronto, ON, Canada; The Arthur and Sonia Labatt Brain Tumour Research Centre, The Hospital for Sick Children, Toronto, ON, Canada.
Betty LuuDevelopmental & Stem Cell Biology Program, The Hospital for Sick Children, Toronto, ON, Canada; The Arthur and Sonia Labatt Brain Tumour Research Centre, The Hospital for Sick Children, Toronto, ON, Canada.
Branden S MoriarityDepartment of Pediatrics, University of Minnesota, Minneapolis, MN 55455, USA; Center for Genome Engineering, University of Minnesota, Minneapolis, MN 55455, USA; Masonic Cancer Center, University of Minnesota, Minneapolis, MN 55455, USA.
Shirin IlkhanizadehDepartment of Neurology and the Helen Diller Family Comprehensive Cancer Center, University of California, San Francisco, San Francisco, CA 94158, USA.
Lukas ChavezHopp-Children's Cancer Center (KiTZ), Heidelberg, Germany; Division of Pediatric Neurooncology, German Cancer Research Center (DKFZ), German Cancer Consortium (DKTK), Heidelberg, Germany.
Chunying YuDevelopmental & Stem Cell Biology Program, The Hospital for Sick Children, Toronto, ON, Canada.
Kathreena M KurianInstitute of Clinical Neurosciences, Level 1, Learning and Research Building, Southmead Hospital, University of Bristol, Bristol BS10 5NB, UK.
Thierry MagnaldoInstitute for Research on Cancer and Aging, Nice UMR CNRS 7284 INSERM U1081 UNS/UCA, Nice, France.
Nicolas SevenetInstitut Bergonie & INSERM U1218, Universite de Bordeaux, 229 cours de l'Argonne, 33076 Bordeaux Cedex, France.
Philipp KochCentral Institute of Mental Health, University of Heidelberg/Medical Faculty Mannheim and Hector Institut for Translational Brain Research (HITBR gGmbH), Mannheim, Germany; German Cancer Research Center (DKFZ), Heidelberg, Germany.
Steven M PollardMRC Centre for Regenerative Medicine and Cancer Research UK Edinburgh Centre, University of Edinburgh, Edinburgh, UK.
Peter DirksDevelopmental & Stem Cell Biology Program, The Hospital for Sick Children, Toronto, ON, Canada; Division of Neurosurgery, The Hospital for Sick Children, Toronto, ON, Canada; The Arthur and Sonia Labatt Brain Tumour Research Centre, The Hospital for Sick Children, Toronto, ON, Canada.
Michael P SnyderDepartment of Genetics, Stanford University School of Medicine, Stanford, CA 94305, USA.
David A LargaespadaDepartment of Pediatrics, University of Minnesota, Minneapolis, MN 55455, USA; Center for Genome Engineering, University of Minnesota, Minneapolis, MN 55455, USA; Masonic Cancer Center, University of Minnesota, Minneapolis, MN 55455, USA.
Yoon Jae ChoDivision of Pediatric Neurology, Department of Pediatrics, Oregon Health & Science University, Portland, OR, USA; Papé Family Pediatric Research Institute, Department of Pediatrics, Oregon Health & Science University, Portland, OR, USA; Knight Cancer Institute, Oregon Health & Science University, Portland, OR, USA.
Joanna J PhillipsDepartments of Neurological Surgery and Pathology, University of California, San Francisco, CA 94158, USA.
Fredrik J SwartlingDepartment of Immunology, Genetics and Pathology, Science for Life Laboratory, Uppsala University, 751 85 Uppsala, Sweden.
A Sorana MorrissyDevelopmental & Stem Cell Biology Program, The Hospital for Sick Children, Toronto, ON, Canada; Department of Biochemistry and Molecular Biology, University of Calgary, Calgary, AB, Canada; Charbonneau Cancer Institute, University of Calgary, Calgary, AB, Canada; Alberta Children's Hospital Research Institute, Calgary, AB, Canada; The Arthur and Sonia Labatt Brain Tumour Research Centre, The Hospital for Sick Children, Toronto, ON, Canada.
Marcel KoolHopp-Children's Cancer Center (KiTZ), Heidelberg, Germany; Division of Pediatric Neurooncology, German Cancer Research Center (DKFZ), German Cancer Consortium (DKTK), Heidelberg, Germany.
Stefan M PfisterHopp-Children's Cancer Center (KiTZ), Heidelberg, Germany; Division of Pediatric Neurooncology, German Cancer Research Center (DKFZ), German Cancer Consortium (DKTK), Heidelberg, Germany; Department of Pediatric Hematology and Oncology, Heidelberg University Hospital, Heidelberg, Germany.
Michael D TaylorDevelopmental & Stem Cell Biology Program, The Hospital for Sick Children, Toronto, ON, Canada; Division of Neurosurgery, The Hospital for Sick Children, Toronto, ON, Canada; The Arthur and Sonia Labatt Brain Tumour Research Centre, The Hospital for Sick Children, Toronto, ON, Canada; Department of Laboratory Medicine and Pathobiology, University of Toronto, Toronto, ON, Canada.
Austin SmithWellcome Trust-MRC Stem Cell Institute, University of Cambridge, Tennis Court Road, Cambridge CB2 1QR, UK.
William A WeissDepartment of Neurology and the Helen Diller Family Comprehensive Cancer Center, University of California, San Francisco, San Francisco, CA 94158, USA; Departments of Pediatrics, Neurosurgery and Brain Tumor Research Center, University of California, San Francisco, San Francisco, CA 94158, USA. Electronic address: waweiss@gmail.com.
University of California, San Francisco · USUniversity of Cambridge · GBHospital for Sick Children · CAUCSF Helen Diller Family Comprehensive Cancer Center · USSickKids Foundation · CAStanford University · USUniversity of Minnesota · USUppsala University · SEGerman Cancer Research Center · DEHeidelberg University · DEHopp Children's Cancer Center Heidelberg · DEInserm · FRInstitut de Recherche sur le Cancer et le Vieillissement de Nice · FRMRC Centre for Regenerative Medicine · GBOregon Health & Science University · USSouthmead Hospital · GBUniversity Hospital Heidelberg · DEUniversity of Calgary · CA
Funding
Translational InformaticsP30CA082103 · NCI · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI Jennifer Leigh Clarke · 1999 to 2026
$209.7MTissue CoreP50CA097257 · NCI · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI Joseph F Costello · 2002 to 2026
$57.4MMEDICAL SCIENTIST TRAINING PROGRAMT32GM007618 · NIGMS · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI ANDERSON, MARK S · 1985 to 2020
$29.6MMolecular and Cellular Mechanisms in CancerT32CA108462 · NCI · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI GOGA, ANDREI · 2004 to 2024
$10.4MMouse models to discover new drivers of medulloblastomaR01CA159859 · NCI · SANFORD BURNHAM PREBYS MEDICAL DISCOVERY INSTITUTE · PI Michael D. Taylor, WILLIAM A WEISS · 2011 to 2026
$10.4MTraining Program in Translational Brain Tumor ResearchT32CA151022 · NCI · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI Joseph F Costello · 2010 to 2026
$6.6MIntegrating targeted and immunotherapy to treat genetically heterogeneous cancersU01CA217864 · NCI · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI BALMAIN, ALLAN, KRUMMEL, MATTHEW F · 2017 to 2021
$5.1MRemodeling the translatome in N-myc mediated medulloblastoma and its therapeutic implicationsR01NS125668 · NINDS · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI Davide Ruggero, WILLIAM A WEISS · 2022 to 2026
$3.3MMYCN, mTOR and translation control in medulloblastomaR01NS089868 · NINDS · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI RUGGERO, DAVIDE, WEISS, WILLIAM A · 2014 to 2018
$3.1MPrevention and treatment of lethal metastases in group 3 medulloblastomaR01NS106155 · NINDS · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI TAYLOR, MICHAEL D., WEISS, WILLIAM A · 2018 to 2022
$2.8MIdentification of novel cooperating partners of MYCN in neuroblastoma tumorigenesisK99CA197484 · NCI · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI HUANG, MILLER · 2016 to 2017
$322kCancer Research UK 17368Cancer Research UK 21992Department of HealthMedical Research Council G1001028Medical Research Council G1100526NCI NIH HHS K99 CA197484NCI NIH HHS P30 CA082103NCI NIH HHS P50 CA097257NCI NIH HHS R01 CA159859NCI NIH HHS T32 CA108462NCI NIH HHS U01 CA217864NIGMS NIH HHS T32 GM007618NINDS NIH HHS R01 NS089868NINDS NIH HHS R01 NS106155NINDS NIH HHS R01 NS125668Wellcome Trust 201511/Z/16/Z
Abstract
Human neural stem cell cultures provide progenitor cells that are potential cells of origin for brain cancers. However, the extent to which genetic predisposition to tumor formation can be faithfully captured in stem cell lines is uncertain. Here, we evaluated neuroepithelial stem (NES) cells, representative of cerebellar progenitors. We transduced NES cells with MYCN, observing medulloblastoma upon orthotopic implantation in mice. Significantly, transcriptomes and patterns of DNA methylation from xenograft tumors were globally more representative of human medulloblastoma compared to a MYCN-driven genetically engineered mouse model. Orthotopic transplantation of NES cells generated from Gorlin syndrome patients, who are predisposed to medulloblastoma due to germline-mutated PTCH1, also generated medulloblastoma. We engineered candidate cooperating mutations in Gorlin NES cells, with mutation of DDX3X or loss of GSE1 both accelerating tumorigenesis. These findings demonstrate that human NES cells provide a potent experimental resource for dissecting genetic causation in medulloblastoma.
Indexed as
AnimalsBasal Cell Nevus SyndromeBrain NeoplasmsCarcinogenesisDEAD-box RNA HelicasesDisease Models, AnimalGenetic EngineeringGenetic Predisposition to DiseaseHumansMedulloblastomaMiceMice, SCIDNeoplasm ProteinsNeural Stem CellsNeuroepithelial CellsN-Myc Proto-Oncogene ProteinDDX3X protein, humanDEAD-box RNA HelicasesGSE1 protein, humanMYCN protein, humanNeoplasm ProteinsN-Myc Proto-Oncogene ProteinPatched-1 ReceptorPTCH1 protein, humanhuman pluripotent stem cellsmedulloblastomaneuroepithelial stem cellsSHH
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