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@ARTICLE{Staffa:127549,
author = {L. Staffa$^*$ and F. Echterdiek$^*$ and N. Nelius$^*$ and
A. Benner$^*$ and W. Werft$^*$ and B. Lahrmann and N. Grabe
and M. Schneider and M. Tariverdian and M. von Knebel
Doeberitz$^*$ and H. Bläker and M. Kloor$^*$},
title = {{M}ismatch repair-deficient crypt foci in {L}ynch
syndrome--molecular alterations and association with
clinical parameters.},
journal = {PLoS one},
volume = {10},
number = {3},
issn = {1932-6203},
address = {Lawrence, Kan.},
publisher = {PLoS},
reportid = {DKFZ-2017-03572},
pages = {e0121980 -},
year = {2015},
abstract = {Lynch syndrome is caused by germline mutations of DNA
mismatch repair (MMR) genes, most frequently MLH1 and MSH2.
Recently, MMR-deficient crypt foci (MMR-DCF) have been
identified as a novel lesion which occurs at high frequency
in the intestinal mucosa from Lynch syndrome mutation
carriers, but very rarely progress to cancer. To shed light
on molecular alterations and clinical associations of
MMR-DCF, we systematically searched the intestinal mucosa
from Lynch syndrome patients for MMR-DCF by
immunohistochemistry. The identified lesions were
characterised for alterations in microsatellite-bearing
genes with proven or suspected role in malignant
transformation. We demonstrate that the prevalence of
MMR-DCF (mean 0.84 MMR-DCF per 1 cm2 mucosa in the
colorectum of Lynch syndrome patients) was significantly
associated with patients' age, but not with patients'
gender. No MMR-DCF were detectable in the mucosa of patients
with sporadic MSI-H colorectal cancer (n = 12).
Microsatellite instability of at least one tested marker was
detected in $89\%$ of the MMR-DCF examined, indicating an
immediate onset of microsatellite instability after MMR gene
inactivation. Coding microsatellite mutations were most
frequent in the genes HT001 (ASTE1) with $33\%,$ followed by
AIM2 $(17\%)$ and BAX $(10\%).$ Though MMR deficiency alone
appears to be insufficient for malignant transformation, it
leads to measurable microsatellite instability even in
single MMR-deficient crypts. Our data indicate for the first
time that the frequency of MMR-DCF increases with patients'
age. Similar patterns of coding microsatellite instability
in MMR-DCF and MMR-deficient cancers suggest that certain
combinations of coding microsatellite mutations, including
mutations of the HT001, AIM2 and BAX gene, may contribute to
the progression of MMR-deficient lesions into MMR-deficient
cancers.},
keywords = {AIM2 protein, human (NLM Chemicals) / ASTE1 protein, human
(NLM Chemicals) / Adaptor Proteins, Signal Transducing (NLM
Chemicals) / BAX protein, human (NLM Chemicals) /
DNA-Binding Proteins (NLM Chemicals) / MLH1 protein, human
(NLM Chemicals) / Nuclear Proteins (NLM Chemicals) /
Proteins (NLM Chemicals) / bcl-2-Associated X Protein (NLM
Chemicals) / MSH2 protein, human (NLM Chemicals) / MutL
Protein Homolog 1 (NLM Chemicals) / MutS Homolog 2 Protein
(NLM Chemicals)},
cin = {C060 / G105},
ddc = {500},
cid = {I:(DE-He78)C060-20160331 / I:(DE-He78)G105-20160331},
pnm = {317 - Translational cancer research (POF3-317)},
pid = {G:(DE-HGF)POF3-317},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:25816162},
pmc = {pmc:PMC4376900},
doi = {10.1371/journal.pone.0121980},
url = {https://inrepo02.dkfz.de/record/127549},
}