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@ARTICLE{Richter:306609,
author = {G. H. S. Richter$^*$ and A. Ranft$^*$ and M. Kerkhoff$^*$
and M. Jens$^*$ and I. E. Kirchberg$^*$ and U. Dirksen$^*$},
title = {{T}he {P}uzzle of {G}enetic {S}tability and {C}hromosomal
{C}opy {N}umber {A}lterations for the {T}herapy of {E}wing
{S}arcoma.},
journal = {Cancers},
volume = {17},
number = {22},
issn = {2072-6694},
address = {Basel},
publisher = {MDPI},
reportid = {DKFZ-2025-02646},
pages = {3719},
year = {2025},
abstract = {Studies of the genomic stability of Ewing sarcoma (EwS)
have produced contradictory findings. While they are
generally characterized by low mutation rates of individual
genes, several cases exhibit genomic alterations that
manifest as chromosomal gains and losses. Taken together,
these alterations represent independent biomarkers for EwS,
such as loss of heterozygosity (LOH) or an altered genome.
Patients with primary EwS tumors with fewer than three copy
number alterations (CNAs) have a better prognosis than those
with more CNAs. The functional mechanisms underlying this
chromosomal instability are not yet clear. However, there
are indications that this may be directly caused by the
EWSR1::ETS translocations that are characteristic of EwS.
The transcriptional behavior of the chimeric transcription
factor EWSR1-FLI1 leads to the formation of R-loop DNA-RNA
hybrids that form when RNA binds back to DNA during
transcription and increased replication stress, which may
result in structural chromosomal changes. Additionally, the
formation of EWSR1 fusion genes in EwS results in the loss
of one or both wild-type EWSR1 alleles in sarcoma cells. As
chromosome segregation has been observed under loss of
wild-type EWSR1, EWSR1 loss has been proposed as a potential
source of LOH. So, it is highly probable that a chromosomal
translocation and the subsequent formation of the EWSR1-ETS
fusion protein cause the genomic alterations in EwS. This
indicates that targeted therapy should be directed against
the CNA and LOH biology caused by the fusion protein.},
subtyp = {Review Article},
keywords = {EWSR1 haploinsufficiency (Other) / Ewing sarcoma (Other) /
biomarker (Other) / loss of heterozygosity (Other) /
replication stress (Other) / replication stress directed
therapy (Other)},
cin = {BE01 / ED01},
ddc = {610},
cid = {I:(DE-He78)BE01-20160331 / I:(DE-He78)ED01-20160331},
pnm = {899 - ohne Topic (POF4-899)},
pid = {G:(DE-HGF)POF4-899},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:41301081},
doi = {10.3390/cancers17223719},
url = {https://inrepo02.dkfz.de/record/306609},
}