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@ARTICLE{Pschl:120154,
author = {J. Pöschl and S. Stark$^*$ and P. Neumann and S.
Gröbner$^*$ and D. Kawauchi$^*$ and D. Jones$^*$ and P. A.
Northcott$^*$ and P. Lichter$^*$ and S. Pfister$^*$ and M.
Kool$^*$ and U. Schüller},
title = {{G}enomic and transcriptomic analyses match medulloblastoma
mouse models to their human counterparts.},
journal = {Acta neuropathologica},
volume = {128},
number = {1},
issn = {1432-0533},
address = {Berlin},
publisher = {Springer},
reportid = {DKFZ-2017-00737},
pages = {123 - 136},
year = {2014},
abstract = {Medulloblastoma is a malignant embryonal brain tumor with
highly variable outcome. In order to study the biology of
this tumor and to perform preclinical treatment studies, a
lot of effort has been put into the generation of
appropriate mouse models. The usage of these models,
however, has become debatable with the advances in human
medulloblastoma subgrouping. This study brings together
multiple relevant mouse models and matches genetic
alterations and gene expression data of 140 murine tumors
with 423 human medulloblastomas in a global way. Using AGDEX
analysis and k-means clustering, we show that the
Blbp-cre::Ctnnb1(ex3)(Fl/+)Trp53 (Fl/Fl) mouse model fits
well to human WNT medulloblastoma, and that, among various
Myc- or Mycn-based mouse medulloblastomas, tumors in
Glt1-tTA::TRE-MYCN/Luc mice proved to be most specific for
human group 3 medulloblastoma. None of the analyzed models
displayed a significant match to group 4 tumors.
Intriguingly, mice with Ptch1 or Smo mutations selectively
modeled SHH medulloblastomas of adulthood, although such
mutations occur in all human age groups. We therefore
suggest that the infantile or adult gene expression pattern
of SHH MBs are not solely determined by specific mutations.
This is supported by the observation that human
medulloblastomas with PTCH1 mutations displayed more
similarities to PTCH1 wild-type tumors of the same age group
than to PTCH1-mutated tumors of the other age group.
Together, we provide novel insights into previously
unrecognized specificity of distinct models and suggest
these findings as a solid basis to choose the appropriate
model for preclinical studies on medulloblastoma.},
keywords = {Hedgehog Proteins (NLM Chemicals) / PTCH protein, human
(NLM Chemicals) / Patched Receptors (NLM Chemicals) /
Patched-1 Receptor (NLM Chemicals) / Ptch1 protein, mouse
(NLM Chemicals) / Receptors, Cell Surface (NLM Chemicals) /
SHH protein, human (NLM Chemicals)},
cin = {B062 / B060},
ddc = {610},
cid = {I:(DE-He78)B062-20160331 / I:(DE-He78)B060-20160331},
pnm = {312 - Functional and structural genomics (POF3-312)},
pid = {G:(DE-HGF)POF3-312},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:24871706},
doi = {10.1007/s00401-014-1297-8},
url = {https://inrepo02.dkfz.de/record/120154},
}