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@ARTICLE{Appay:131488,
author = {R. Appay and N. Macagno and L. Padovani and A.
Korshunov$^*$ and M. Kool$^*$ and N. André and D. Scavarda
and T. Pietsch and D. Figarella-Branger},
title = {{HGNET}-{BCOR} {T}umors of the {C}erebellum:
{C}linicopathologic and {M}olecular {C}haracterization of 3
{C}ases.},
journal = {The American journal of surgical pathology},
volume = {41},
number = {9},
issn = {0147-5185},
address = {Philadelphia, Pa.},
publisher = {Lippincott Williams $\&$ Wilkins},
reportid = {DKFZ-2017-06155},
pages = {1254 - 1260},
year = {2017},
abstract = {The central nervous system (CNS) high-grade neuroepithelial
tumor with BCOR alteration (CNS HGNET-BCOR) is a recently
described molecular entity. We report 3 new CNS HGNET-BCOR
cases sharing common clinical presentation and pathologic
features. The 3 cases concerned children aged 3 to 7 years
who presented with a voluminous mass of the cerebellum.
Pathologic features included proliferation of uniform
spindle to ovoid cells with fine chromatin associated with a
rich arborizing capillary network. Methylation profiling
classified these cases as CNS HGNET-BCOR tumors. Polymerase
chain reaction analysis confirmed the presence of internal
tandem duplications in the C-terminus of BCOR (BCOR-ITD), a
characteristic of these tumors, in all 3 cases.
Immunohistochemistry showed a strong nuclear BCOR
expression. In 2 cases, local recurrence occurred within 6
months. The third case, a patient who received a
craniospinal irradiation after total surgical removal
followed by a metronomics maintenance with irinotecan,
temozolomide, and itraconazole, is still free of disease 14
months after diagnosis. In summary, CNS HGNET-BCOR
represents a rare tumor occurring in young patients with
dismal prognosis. BCOR nuclear immunoreactivity is highly
suggestive of a BCOR-ITD. Whether CNS HGNET-BCOR should be
classified among the category of 'embryonal tumors' or
within the category of 'mesenchymal, nonmeningothelial
tumors' remains to be clarified. Because CNS HGNET-BCOR
share pathologic features and characteristic BCOR-ITD with
clear cell sarcoma of the kidney, these tumors may represent
local variants of the same entity.},
keywords = {BCOR protein, human (NLM Chemicals) / Proto-Oncogene
Proteins (NLM Chemicals) / Repressor Proteins (NLM
Chemicals)},
cin = {G380 / B062 / L101},
ddc = {610},
cid = {I:(DE-He78)G380-20160331 / I:(DE-He78)B062-20160331 /
I:(DE-He78)L101-20160331},
pnm = {319H - Addenda (POF3-319H)},
pid = {G:(DE-HGF)POF3-319H},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:28704208},
doi = {10.1097/PAS.0000000000000866},
url = {https://inrepo02.dkfz.de/record/131488},
}