% IMPORTANT: The following is UTF-8 encoded. This means that in the presence
% of non-ASCII characters, it will not work with BibTeX 0.99 or older.
% Instead, you should use an up-to-date BibTeX implementation like “bibtex8” or
% “biber”.
@ARTICLE{Huang:144001,
author = {M. Huang and J. Tailor and Q. Zhen and A. H. Gillmor and M.
L. Miller and H. Weishaupt and J. Chen and T. Zheng and E.
K. Nash and L. K. McHenry and Z. An and F. Ye and Y.
Takashima and J. Clarke and H. Ayetey and F. M. G. Cavalli
and B. Luu and B. S. Moriarity and S. Ilkhanizadeh and L.
Chavez$^*$ and C. Yu and K. M. Kurian and T. Magnaldo and N.
Sevenet and P. Koch$^*$ and S. M. Pollard and P. Dirks and
M. P. Snyder and D. A. Largaespada and Y. J. Cho and J. J.
Phillips and F. J. Swartling and A. S. Morrissy and M.
Kool$^*$ and S. M. Pfister$^*$ and M. D. Taylor and A. Smith
and W. A. Weiss},
title = {{E}ngineering {G}enetic {P}redisposition in {H}uman
{N}euroepithelial {S}tem {C}ells {R}ecapitulates
{M}edulloblastoma {T}umorigenesis.},
journal = {Cell stem cell},
volume = {25},
number = {3},
issn = {1934-5909},
address = {Amsterdam [u.a.]},
publisher = {Elsevier},
reportid = {DKFZ-2019-01552},
pages = {433-446.e7},
year = {2019},
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.},
cin = {B062},
ddc = {570},
cid = {I:(DE-He78)B062-20160331},
pnm = {312 - Functional and structural genomics (POF3-312)},
pid = {G:(DE-HGF)POF3-312},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:31204176},
doi = {10.1016/j.stem.2019.05.013},
url = {https://inrepo02.dkfz.de/record/144001},
}