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@ARTICLE{Siegfried:142875,
author = {A. Siegfried and A. Rousseau and C.-A. Maurage and S.
Pericart and Y. Nicaise and F. Escudie and D. Grand and A.
Delrieu and A. Gomez-Brouchet and S. Le Guellec and C.
Franchet and S. Boetto and M. Vinchon and J.-C. Sol and
F.-E. Roux and V. Rigau and A.-I. Bertozzi and D. Jones$^*$
and D. Figarella-Branger and E. Uro-Coste},
title = {{EWSR}1-{PATZ}1 gene fusion may define a new glioneuronal
tumor entity.},
journal = {Brain pathology},
volume = {29},
number = {1},
issn = {1015-6305},
address = {Oxford},
publisher = {Wiley-Blackwell},
reportid = {DKFZ-2019-00505},
pages = {53 - 62},
year = {2019},
abstract = {We investigated the challenging diagnostic case of a
ventricular cystic glioneuronal tumor with papillary
features, by RNA sequencing using the Illumina TruSight RNA
Fusion panel. We did not retrieve the SLC44A1-PRKCA fusion
gene specific for papillary glioneuronal tumor, but an
EWSR1-PATZ1 fusion transcript. RT-PCR followed by Sanger
sequencing confirmed the EWSR1-PATZ1 fusion. It matched with
canonic EWSR1 fusion oncogene, juxtaposing the entire
N-terminal transcriptional activation domain of EWSR1 gene
and the C-terminal DNA binding domain of a transcription
factor gene, PATZ1. PATZ1 protein belongs to the BTB-ZF
(broad-complex, tramtrack and bric-à-brac -zinc finger)
family. It directly regulates Pou5f1 and Nanog and is
essential to maintaining stemness by inhibiting neural
differentiation. EWSR1-PATZ1 fusion is a rare event in
tumors: it was only reported in six round cell sarcomas and
in three gliomas of three exclusively molecular studies. The
first reported glioma was a BRAFV600E negative
ganglioglioma, the second a BRAFV600E negative glioneuronal
tumor, not otherwise specified and the third, very recently
reported, a high grade glioma, not otherwise specified. In
our study, forty BRAFV600E negative gangliogliomas were
screened by FISH using EWSR1 break-apart probes. We
performed methylation profiling for the index case and for
seven out of the ten FISH positive cases. The index case
clustered apart from other pediatric low grade glioneuronal
entities, and specifically from the well-defined
ganglioglioma methylation group. An additional pediatric
intraventricular ganglioglioma clustered slightly more
closely with ganglioglioma, but showed differences from the
main ganglioglioma group and similarities with the index
case. Both cases harbored copy number variations at the
PATZ1 locus. EWSR1-PATZ1 gene fusion might define a new type
of glioneuronal tumors, distinct from gangliogliomas.},
cin = {B360 / L101},
ddc = {610},
cid = {I:(DE-He78)B360-20160331 / I:(DE-He78)L101-20160331},
pnm = {312 - Functional and structural genomics (POF3-312)},
pid = {G:(DE-HGF)POF3-312},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:29679497},
doi = {10.1111/bpa.12619},
url = {https://inrepo02.dkfz.de/record/142875},
}