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@ARTICLE{Upadhyaya:168085,
author = {S. A. Upadhyaya and G. Robinson and A. Onar-Thomas and B.
A. Orr and P. Johann and G. Wu and C. A. Billups and R. G.
Tatevossian and S. K. Dhanda and A. Srinivasan and A.
Broniscer and I. Qaddoumi and A. Vinitsky and G. T.
Armstrong and A. Bendel and T. E. Hassall and S. Partap and
P. G. Fisher and J. R. Crawford and M. M. Chintagumpala and
E. Bouffet and S. Gururangan and R. Mostafavi and R. P.
Sanders and P. Klimo and Z. Patay and D. J. Indelicato and
K. E. Nichols and F. A. Boop and T. E. Merchant and M.
Kool$^*$ and D. W. Ellison and A. Gajjar},
title = {{R}elevance of {M}olecular {G}roups in {C}hildren with
{N}ewly {D}iagnosed {A}typical {T}eratoid {R}habdoid
{T}umor: {R}esults from {P}rospective {S}t. {J}ude
{M}ulti-{I}nstitutional {T}rials.},
journal = {Clinical cancer research},
volume = {27},
number = {10},
issn = {1557-3265},
address = {Philadelphia, Pa. [u.a.]},
publisher = {AACR},
reportid = {DKFZ-2021-00673},
pages = {2879-2889},
year = {2021},
note = {27(10):2879-2889},
abstract = {Report relevance of molecular groups to clinico-pathologic
features, germline SMARCB1/SMARCA4 alterations (GLA), and
survival of children with atypical teratoid rhabdoid tumor
(ATRT) treated in two multi-institutional clinical
trials.Seventy-four participants with newly diagnosed ATRT
were treated in two trials: infants (SJYC07: age<3 years;
n=52) and children (SJMB03: age 3-21 years; n=22), using
surgery, conventional chemotherapy (infants), or dose-dense
chemotherapy with autologous stem cell rescue (children),
and age- and risk-adapted radiation therapy [focal (infants)
and craniospinal (CSI) (children)]. Molecular groups
ATRT-MYC (MYC), ATRT-SHH (SHH), and ATRT-TYR (TYR) were
determined from tumor DNA methylation profiles.Twenty-four
participants $(32\%)$ were alive at time of analysis at a
median follow-up of 8.4 years (range, 3.1-14.1 years).
Methylation profiling classified 64 ATRTs as TYR (n=21), SHH
(n=30), and MYC (n=13), SHH group being associated with
metastatic disease. Among infants, TYR group had the best
overall survival (OS) (P=0.02). However, outcomes did not
differ by molecular groups among infants with non-metastatic
(M0) disease. Children with M0 disease and <1.5 cm2 residual
tumor had a 5-year progression-free survival (PFS) of
72.7{plus $minus}12.7\%$ and OS of 81.8{plus $minus}11\%.$
Infants with M0 disease had a 5-year PFS of 39.1{plus
$minus}11.5\%$ and OS of 51.8{plus $minus}12\%.$ Those with
metastases fared poorly [5-year OS 25{plus $minus}12.5\%$
(children) and $0\%$ (infants)]. SMARCB1 GLAs were not
associated with PFS.Among infants, those with ATRT-TYR had
the best OS. ATRT-SHH was associated with metastases and
consequently with inferior outcomes. Children with
non-metastatic ATRT benefit from post-operative CSI and
adjuvant chemotherapy.},
cin = {B062},
ddc = {610},
cid = {I:(DE-He78)B062-20160331},
pnm = {312 - Funktionelle und strukturelle Genomforschung
(POF4-312)},
pid = {G:(DE-HGF)POF4-312},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:33737307},
doi = {10.1158/1078-0432.CCR-20-4731},
url = {https://inrepo02.dkfz.de/record/168085},
}