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@ARTICLE{Gojo:128032,
author = {J. Gojo and D. Lötsch and S. Spiegl-Kreinecker and K.
Pajtler$^*$ and K. Neumayer and P. Korbel and A. Araki and
A. Brandstetter and T. Mohr and V. Hovestadt$^*$ and L.
Chavez$^*$ and D. Kirchhofer and G. Ricken and H. Stefanits
and A. Korshunov$^*$ and S. Pfister$^*$ and K. Dieckmann and
A. A. Azizi and T. Czech and M. Filipits and M. Kool$^*$ and
A. Peyrl and I. Slavc and W. Berger and C. Haberler},
title = {{T}elomerase activation in posterior fossa group {A}
ependymomas is associated with dismal prognosis and
chromosome 1q gain.},
journal = {Neuro-Oncology},
volume = {19},
number = {9},
issn = {1523-5866},
address = {Oxford},
publisher = {Oxford Univ. Press},
reportid = {DKFZ-2017-04054},
pages = {1183 - 1194},
year = {2017},
abstract = {Ependymomas account for up to $10\%$ of childhood CNS
tumors and have a high rate of tumor recurrence despite
gross total resection. Recently, classification into
molecular ependymoma subgroups has been established, but the
mechanisms underlying the aggressiveness of certain subtypes
remain widely enigmatic. The aim of this study was to
dissect the clinical and biological role of telomerase
reactivation, a frequent mechanism of cancer cells to evade
cellular senescence, in pediatric ependymoma.We determined
telomerase enzymatic activity, hTERT mRNA expression,
promoter methylation, and the rs2853669 single nucleotide
polymorphism located in the hTERT promoter in a
well-characterized cohort of pediatric intracranial
ependymomas.In posterior fossa ependymoma group A (PF-EPN-A)
tumors, telomerase activity varied and was significantly
associated with dismal overall survival, whereas telomerase
reactivation was present in all supratentorial RelA
fusion-positive (ST-EPN-RELA) ependymomas. In silico
analysis of methylation patterns showed that only these two
subgroups harbor hypermethylated hTERT promoters suggesting
telomerase reactivation via epigenetic mechanisms.
Furthermore, chromosome 1q gain, a well-known negative
prognostic factor, was strongly associated with telomerase
reactivation in PF-EPN-A. Additional in silico analyses of
gene expression data confirmed this finding and further
showed enrichment of the E-twenty-six factor, Myc, and E2F
target genes in 1q gained ependymomas. Additionally, 1q
gained tumors showed elevated expression of ETV3, an
E-twenty-six factor gene located on chromosome 1q.Taken
together we describe a subgroup-specific impact of
telomerase reactivation on disease progression in pediatric
ependymoma and provide preliminary evidence for the involved
molecular mechanisms.},
cin = {B062 / G380 / B060 / L101},
ddc = {610},
cid = {I:(DE-He78)B062-20160331 / I:(DE-He78)G380-20160331 /
I:(DE-He78)B060-20160331 / I:(DE-He78)L101-20160331},
pnm = {319H - Addenda (POF3-319H)},
pid = {G:(DE-HGF)POF3-319H},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:28371821},
pmc = {pmc:PMC5570194},
doi = {10.1093/neuonc/nox027},
url = {https://inrepo02.dkfz.de/record/128032},
}