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@ARTICLE{Bolduan:301583,
author = {F. Bolduan and N. Müller-Bötticher and O. Debnath and I.
Eichhorn and Y. Giesecke and A. Wetzel and S. Sahay and T.
Zemojtel and M. Jaeger and U. Ungethuem and C. Roderburg and
C. A. Kunze and A. Lehmann and D. Horst$^*$ and F. Tacke and
R. Eils and B. Wiedenmann and M. Sigal and N. Ishaque},
title = {{S}mall intestinal neuroendocrine tumors lack early genomic
drivers, acquire {DNA} repair defects and harbor hallmarks
of low {REST} expression.},
journal = {Scientific reports},
volume = {15},
number = {1},
issn = {2045-2322},
address = {[London]},
publisher = {Springer Nature},
reportid = {DKFZ-2025-01091},
pages = {17969},
year = {2025},
abstract = {The tumorigenesis of small intestinal neuroendocrine tumors
(siNETs) is not understood and comprehensive genomic and
transcriptomic data sets are limited. Therefore, we
performed whole genome and transcriptome analysis of 39 well
differentiated siNET samples. Our genomic data revealed a
lack of recurrent driver mutations and demonstrated that
multifocal siNETs from individual patients can arise
genetically independently. We detected germline mutations in
Fanconi anemia DNA repair pathway (FANC) genes, involved in
homologous recombination (HR) DNA repair, in $9\%$ of
patients and found mutational signatures of defective HR DNA
repair in late-stage tumor evolution. Furthermore,
transcriptomic analysis revealed low expression of the
transcriptional repressor REST. Summarizing, we identify a
novel common transcriptomic signature of siNETs and
demonstrate that genomic alterations alone do not explain
initial tumor formation, while impaired DNA repair likely
contributes to tumor evolution and represents a potential
pharmaceutical target in a subset of patients.},
keywords = {Humans / Neuroendocrine Tumors: genetics / Neuroendocrine
Tumors: pathology / Neuroendocrine Tumors: metabolism /
Intestinal Neoplasms: genetics / Intestinal Neoplasms:
pathology / DNA Repair: genetics / Gene Expression
Regulation, Neoplastic / Female / Male / Repressor Proteins:
genetics / Repressor Proteins: metabolism / Intestine,
Small: pathology / Intestine, Small: metabolism / Gene
Expression Profiling / Middle Aged / Transcriptome /
Germ-Line Mutation / Genomics / Adult / Repressor Proteins
(NLM Chemicals)},
cin = {BE01},
ddc = {600},
cid = {I:(DE-He78)BE01-20160331},
pnm = {899 - ohne Topic (POF4-899)},
pid = {G:(DE-HGF)POF4-899},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:40410286},
pmc = {pmc:PMC12102166},
doi = {10.1038/s41598-025-01912-4},
url = {https://inrepo02.dkfz.de/record/301583},
}