% 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{Baroni:167455,
author = {L. Baroni and L. Sundaresan and A. Heled and H. Coltin and
K. W. Pajtler$^*$ and T. Lin and T. E. Merchant and R.
McLendon and C. Faria and M. Buntine and C. L. White and S.
M. Pfister$^*$ and M. R. Gilbert and T. S. Armstrong and E.
Bouffet and S. Kumar and M. D. Taylor and K. D. Aldape and
D. W. Ellison and N. G. Gottardo and M. Kool$^*$ and A.
Korshunov$^*$ and J. R. Hansford and V. Ramaswamy},
title = {{U}ltra high-risk {PFA} ependymoma is characterized by loss
of chromosome 6q.},
journal = {Neuro-Oncology},
volume = {23},
number = {8},
issn = {1523-5866},
address = {Oxford},
publisher = {Oxford Univ. Press},
reportid = {DKFZ-2021-00359},
pages = {1360-1370},
year = {2021},
note = {2021 Aug 2;23(8):1360-1370},
abstract = {Within PF-EPN-A, 1q gain is a marker of poor prognosis,
however, it is unclear if within PF-EPN-A additional
cytogenetic events exist which can refine risk
stratification.Five independent non-overlapping cohorts of
PF-EPN-A were analyzed applying genome wide methylation
arrays for chromosomal and clinical variables predictive of
survival.Across all cohorts, 663 PF-EPN-A were identified.
The most common broad copy number event was 1q gain
$(18.9\%),$ followed by 6q loss $(8.6\%),$ 9p gain
$(6.5\%),$ and 22q loss $(6.8\%).$ Within 1q gain tumors,
there was significant enrichment for 6q loss $(17.7\%),$ 10q
loss $(16.9\%)$ and 16q loss $(15.3\%).$ The 5-year
progression free survival (PFS) was strikingly worse in
those patients with 6q loss, with a 5-year PFS of $50\%$
$(95\%CI$ $45-55\%)$ for balanced tumors, compared with
$32\%$ $(95\%CI$ $24-44\%)$ for 1q gain only, $7.3\%$
$(95\%CI$ $2.0-27\%)$ for 6q loss only and 0 for both 1q
gain and 6q loss (p=1.65x10 -13 ). After accounting for
treatment, 6q loss remained the most significant independent
predictor of survival in PF-EPN-A but is not in PF-EPN-B.
Distant relapses were more common in 1q gain irrespective of
6q loss. RNA-sequencing comparing 6q loss to 6q balanced
PF-EPN-A suggests that 6q loss forms a biologically distinct
group.We have identified an ultra high-risk PF-EPN-A
ependymoma subgroup, which can be reliably ascertained using
cytogenetic markers in routine clinical use. A change in
treatment paradigm is urgently needed for this particular
subset of PF-EPN-A where novel therapies should be
prioritized for upfront therapy.},
keywords = {1q gain (Other) / 6qloss (Other) / PFA (Other) / PFB
(Other) / ependymoma (Other) / posterior fossa (Other) /
subgrouping (Other)},
cin = {B062 / HD01 / B300},
ddc = {610},
cid = {I:(DE-He78)B062-20160331 / I:(DE-He78)HD01-20160331 /
I:(DE-He78)B300-20160331},
pnm = {312 - Funktionelle und strukturelle Genomforschung
(POF4-312)},
pid = {G:(DE-HGF)POF4-312},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:33580238},
doi = {10.1093/neuonc/noab034},
url = {https://inrepo02.dkfz.de/record/167455},
}