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@ARTICLE{Wagner:162928,
author = {Wagner, Matias and Lorenz, Georg and Volk, Alexander E and
Brunet, Theresa and Edbauer, Dieter and Berutti, Riccardo
and Zhao, Chen and Anderl-Straub, Sarah and Bertram, Lars
and Danek, Adrian and Deschauer, Marcus and Dill, Veronika
and Fassbender, Klaus and Fliessbach, Klaus and Götze,
Katharina S and Jahn, Holger and Kornhuber, Johannes and
Landwehrmeyer, Bernhard and Lauer, Martin and Obrig,
Hellmuth and Prudlo, Johannes and Schneider, Anja and
Schroeter, Matthias L and Uttner, Ingo and Vukovich, Ruth C.
and Wiltfang, Jens and Winkler, Andrea S and Zhou, Qihui and
Ludolph, Albert C and Oexle, Konrad and Otto, Markus and
Diehl-Schmid, Janine and Winkelmann, Juliane},
collaboration = {consortium, German FTLD},
title = {{C}linico-genetic findings in 509 frontotemporal dementia
patients.},
journal = {Molecular psychiatry},
volume = {26},
number = {10},
issn = {1476-5578},
address = {London},
publisher = {Macmillan},
reportid = {DZNE-2021-01580},
pages = {5824-5832},
year = {2021},
note = {CC BY},
abstract = {Frontotemporal dementia (FTD) is a clinically and
genetically heterogeneous disorder. To which extent genetic
aberrations dictate clinical presentation remains elusive.
We investigated the spectrum of genetic causes and assessed
the genotype-driven differences in biomarker profiles,
disease severity and clinical manifestation by recruiting
509 FTD patients from different centers of the German FTLD
consortium where individuals were clinically assessed
including biomarker analysis. Exome sequencing as well as
C9orf72 repeat analysis were performed in all patients.
These genetic analyses resulted in a diagnostic yield of
$18.1\%.$ Pathogenic variants in C9orf72 (n = 47), GRN (n =
26), MAPT (n = 11), TBK1 (n = 5), FUS (n = 1), TARDBP (n =
1), and CTSF (n = 1) were identified across all clinical
subtypes of FTD. TBK1-associated FTD was frequent accounting
for $5.4\%$ of solved cases. Detection of a homozygous
missense variant verified CTSF as an FTD gene. ABCA7 was
identified as a candidate gene for monogenic FTD. The
distribution of APOE alleles did not differ significantly
between FTD patients and the average population. Male sex
was weakly associated with clinical manifestation of the
behavioral variant of FTD. Age of onset was lowest in MAPT
patients. Further, high CSF neurofilament light chain levels
were found to be related to GRN-associated FTD. Our study
provides large-scale retrospective clinico-genetic data such
as on disease manifestation and progression of FTD. These
data will be relevant for counseling patients and their
families.},
keywords = {C9orf72 Protein: genetics / Frontotemporal Dementia:
genetics / Genotype / Humans / Male / Mutation /
Retrospective Studies / Exome Sequencing / tau Proteins:
genetics},
cin = {AG Edbauer / AG Schneider / AG Teipel / AG Wiltfang /
Clinical Study Center Ulm / Patient Studies Bonn / AG Zhou},
ddc = {610},
cid = {I:(DE-2719)1110004 / I:(DE-2719)1011305 /
I:(DE-2719)1510100 / I:(DE-2719)1410006 / I:(DE-2719)5000077
/ I:(DE-2719)1011101 / I:(DE-2719)5000080},
pnm = {353 - Clinical and Health Care Research (POF4-353) / 352 -
Disease Mechanisms (POF4-352)},
pid = {G:(DE-HGF)POF4-353 / G:(DE-HGF)POF4-352},
typ = {PUB:(DE-HGF)16},
pmc = {pmc:PMC8758482},
pubmed = {pmid:34561610},
doi = {10.1038/s41380-021-01271-2},
url = {https://pub.dzne.de/record/162928},
}