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@ARTICLE{Sievers:144145,
author = {P. Sievers$^*$ and R. Appay and D. Schrimpf$^*$ and D.
Stichel$^*$ and D. E. Reuss$^*$ and A. K. Wefers$^*$ and A.
Reinhardt$^*$ and R. Coras and V. C. Ruf and S. Schmid and
K. de Stricker and H. B. Boldt and B. W. Kristensen and J.
K. Petersen and B. P. Ulhøi and M. Gardberg and E. Aronica
and M. Hasselblatt and W. Brück and F. Bielle and K.
Mokhtari and B. Lhermitte and W. Wick$^*$ and C.
Herold-Mende and D. Hänggi and S. Brandner and F.
Giangaspero and D. Capper and E. Rushing and P. Wesseling
and S. M. Pfister$^*$ and D. Figarella-Branger and A. von
Deimling$^*$ and F. Sahm$^*$ and D. Jones$^*$},
title = {{R}osette-forming glioneuronal tumors share a distinct
{DNA} methylation profile and mutations in {FGFR}1, with
recurrent co-mutation of {PIK}3{CA} and {NF}1.},
journal = {Acta neuropathologica},
volume = {138},
number = {3},
issn = {1432-0533},
address = {Heidelberg},
publisher = {Springer},
reportid = {DKFZ-2019-01694},
pages = {497-504},
year = {2019},
abstract = {Rosette-forming glioneuronal tumor (RGNT) is a rare brain
neoplasm that primarily affects young adults. Although
alterations affecting the mitogen-activated protein kinase
(MAPK) and phosphoinositide 3-kinase (PI3K) signaling
pathway have been associated with this low-grade entity,
comprehensive molecular investigations of RGNT in larger
series have not been performed to date, and an integrated
view of their genetic and epigenetic profiles is still
lacking. Here we describe a genome-wide DNA methylation and
targeted sequencing-based characterization of a molecularly
distinct class of tumors (n = 30), initially identified
through genome-wide DNA methylation screening among a cohort
of > 30,000 tumors, of which most were diagnosed
histologically as RGNT. FGFR1 hotspot mutations were
observed in all tumors analyzed, with co-occurrence of
PIK3CA mutations in about two-thirds of the cases $(63\%).$
Additional loss-of-function mutations in the tumor
suppressor gene NF1 were detected in a subset of cases
$(33\%).$ Notably, in contrast to most other low-grade
gliomas, these tumors often displayed co-occurrence of two
or even all three of these mutations. Our data highlight
that molecularly defined RGNTs are characterized by highly
recurrent combined genetic alterations affecting both MAPK
and PI3K signaling pathways. Thus, these two pathways appear
to synergistically interact in the formation of RGNT, and
offer potential therapeutic targets for this disease.},
cin = {B300 / B320 / B062 / B360},
ddc = {610},
cid = {I:(DE-He78)B300-20160331 / I:(DE-He78)B320-20160331 /
I:(DE-He78)B062-20160331 / I:(DE-He78)B360-20160331},
pnm = {312 - Functional and structural genomics (POF3-312)},
pid = {G:(DE-HGF)POF3-312},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:31250151},
doi = {10.1007/s00401-019-02038-4},
url = {https://inrepo02.dkfz.de/record/144145},
}