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@ARTICLE{Dutzmann:144822,
author = {C. M. Dutzmann and J. Vogel and C. P. Kratz and K. W.
Pajtler$^*$ and S. M. Pfister$^*$ and B. B. Dörgeloh},
title = {[{U}pdate on {L}i-{F}raumeni syndrome].},
journal = {Der Pathologe},
volume = {40},
number = {6},
issn = {1432-1963},
address = {New York},
publisher = {Springer},
reportid = {DKFZ-2019-02247},
pages = {592-599},
year = {2019},
abstract = {The Li-Fraumeni syndrome (LFS, online Mendelian inheritance
in man, OMIM #151623) is considered to be one of the
currently known most aggressive cancer predisposition
syndromes. The heterogeneous spectrum of tumors is dominated
by bone and soft tissue sarcomas, various brain tumors,
premenopausal breast cancer and adrenocortical carcinoma
(ACC). Even in childhood the cancer risk is very strongly
increased and it is not uncommon for people with LFS to
develop synchronous and metachronous tumors. Typical
histopathological findings and molecular genetic signatures
can help towards the diagnosis. Inheritance is autosomal
dominant and the penetrance appears to be more variable than
previously thought. The prevalence of LFS is approximately
1:5000 with a high interregional variance. The LFS is
caused by germline mutations in the TP53 gene coding for the
protein p53, an essential cellular transcription factor that
initiates antitumor responses to cellular stress, such as
DNA damage. In people with LFS, due to the loss of
functional p53, the protective mechanism of the cells is
weakened resulting in a significantly increased cancer
risk. In order to improve the survival of people with LFS,
structured tumor early recognition and surveillance
strategies are recommended; however, national and
international longitudinal observational studies are needed
to evaluate the cost-effort-benefit balance. For this
reason, the authors have established the LFS cancer
predisposition registry in which all patients with LFS and
other syndromes predisposing to cancer can be registered.
Detailed information can be found at
www.cancer-predisposition.org .},
subtyp = {Review Article},
cin = {B062 / L101},
ddc = {610},
cid = {I:(DE-He78)B062-20160331 / I:(DE-He78)L101-20160331},
pnm = {312 - Functional and structural genomics (POF3-312)},
pid = {G:(DE-HGF)POF3-312},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:31511974},
doi = {10.1007/s00292-019-00657-y},
url = {https://inrepo02.dkfz.de/record/144822},
}