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@ARTICLE{Wang:141144,
author = {A. C. Wang and D. Jones$^*$ and I. J. Abecassis and B. L.
Cole and S. E. S. Leary and C. M. Lockwood and L. Chavez$^*$
and D. Capper$^*$ and A. Korshunov$^*$ and A. Fallah and S.
Wang and C. Ene and J. M. Olson and J. R. Geyer and E. C.
Holland and A. Lee and R. G. Ellenbogen and J. G. Ojemann},
title = {{D}esmoplastic {I}nfantile {G}anglioglioma/{A}strocytoma
({DIG}/{DIA}) {A}re {D}istinct {E}ntities with {F}requent
{BRAFV}600 {M}utations.},
journal = {Molecular cancer research},
volume = {16},
number = {10},
issn = {1541-7786},
address = {Philadelphia, Pa.},
publisher = {AACR},
reportid = {DKFZ-2018-01675},
pages = {1491 - 1498},
year = {2018},
abstract = {Desmoplastic infantile ganglioglioma (DIG) and desmoplastic
infantile astrocytoma (DIA) are extremely rare tumors that
typically arise in infancy; however, these entities have not
been well characterized in terms of genetic alterations or
clinical outcomes. Here, through a multi-institutional
collaboration, the largest cohort of DIG/DIA to date is
examined using advanced laboratory and data processing
techniques. Targeted DNA exome sequencing and DNA
methylation profiling were performed on tumor specimens
obtained from different patients (n = 8) diagnosed
histologically as DIG/DIGA. Two of these cases clustered
with other tumor entities, and were excluded from analysis.
The remaining 16 cases were confirmed to be DIG/DIA by
histology and by DNA methylation profiling. Somatic BRAF
gene mutations were discovered in 7 instances $(43.8\%);$ 4
were BRAFV600E mutations, and 3 were BRAFV600D mutations.
Three instances of malignant transformation were found, and
sequencing of the recurrence demonstrated a new TP53
mutation in one case, new ATRX deletion in one case, and in
the third case, the original tumor harbored an EML4-ALK
fusion, also present at recurrence. DIG/DIA are distinct
pathologic entities that frequently harbor BRAFV600
mutations. Complete surgical resection is the ideal
treatment, and overall prognosis is excellent. While, the
small sample size and incomplete surgical records limit a
definitive conclusion about the risk of tumor recurrence,
the risk appears quite low. In rare cases with wild-type
BRAF, malignant progression can be observed, frequently with
the acquisition of other genetic alterations.Implications:
DIG/DIA are a distinct molecular entity, with a subset
frequently harboring either BRAFV600E or BRAFV600D
mutations. Mol Cancer Res; 16(10); 1491-8. ©2018 AACR.},
cin = {B062 / G380},
ddc = {610},
cid = {I:(DE-He78)B062-20160331 / I:(DE-He78)G380-20160331},
pnm = {319H - Addenda (POF3-319H)},
pid = {G:(DE-HGF)POF3-319H},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:30006355},
doi = {10.1158/1541-7786.MCR-17-0507},
url = {https://inrepo02.dkfz.de/record/141144},
}