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@ARTICLE{Dormann:136697,
author = {Dormann, Dorothee and Madl, Tobias and Valori, Chiara F and
Bentmann, Eva and Tahirovic, Sabina and Abou-Ajram, Claudia
and Kremmer, Elisabeth and Ansorge, Olaf and Mackenzie, Ian
R A and Neumann, Manuela and Haass, Christian},
title = {{A}rginine methylation next to the {PY}-{NLS} modulates
{T}ransportin binding and nuclear import of {FUS}.},
journal = {The EMBO journal},
volume = {31},
number = {22},
issn = {0261-4189},
address = {Hoboken, NJ [u.a.]},
publisher = {Wiley},
reportid = {DZNE-2020-03019},
pages = {4258-4275},
year = {2012},
abstract = {Fused in sarcoma (FUS) is a nuclear protein that carries a
proline-tyrosine nuclear localization signal (PY-NLS) and is
imported into the nucleus via Transportin (TRN). Defects in
nuclear import of FUS have been implicated in
neurodegeneration, since mutations in the PY-NLS of FUS
cause amyotrophic lateral sclerosis (ALS). Moreover, FUS is
deposited in the cytosol in a subset of frontotemporal lobar
degeneration (FTLD) patients. Here, we show that arginine
methylation modulates nuclear import of FUS via a novel
TRN-binding epitope. Chemical or genetic inhibition of
arginine methylation restores TRN-mediated nuclear import of
ALS-associated FUS mutants. The unmethylated
arginine-glycine-glycine domain preceding the PY-NLS
interacts with TRN and arginine methylation in this domain
reduces TRN binding. Inclusions in ALS-FUS patients contain
methylated FUS, while inclusions in FTLD-FUS patients are
not methylated. Together with recent findings that FUS
co-aggregates with two related proteins of the FET family
and TRN in FTLD-FUS but not in ALS-FUS, our study provides
evidence that these two diseases may be initiated by
distinct pathomechanisms and implicates alterations in
arginine methylation in pathogenesis.},
keywords = {Active Transport, Cell Nucleus / Amino Acid Sequence /
Amyotrophic Lateral Sclerosis: genetics / Amyotrophic
Lateral Sclerosis: metabolism / Arginine: metabolism / Cell
Nucleus: metabolism / Frontotemporal Lobar Degeneration:
metabolism / Gene Silencing / HeLa Cells / Humans /
Karyopherins: genetics / Karyopherins: metabolism /
Methylation / Molecular Sequence Data / Nuclear Localization
Signals: metabolism / Proline: metabolism / Protein Binding
/ Protein-Arginine N-Methyltransferases: genetics /
Protein-Arginine N-Methyltransferases: metabolism /
RNA-Binding Protein FUS: genetics / RNA-Binding Protein FUS:
metabolism / Repressor Proteins: genetics / Repressor
Proteins: metabolism / Signal Transduction / Tyrosine:
metabolism / Karyopherins (NLM Chemicals) / Nuclear
Localization Signals (NLM Chemicals) / RNA-Binding Protein
FUS (NLM Chemicals) / Repressor Proteins (NLM Chemicals) /
Tyrosine (NLM Chemicals) / Arginine (NLM Chemicals) /
Proline (NLM Chemicals) / PRMT1 protein, human (NLM
Chemicals) / Protein-Arginine N-Methyltransferases (NLM
Chemicals)},
cin = {AG Neumann},
ddc = {570},
cid = {I:(DE-2719)1210003},
pnm = {344 - Clinical and Health Care Research (POF3-344)},
pid = {G:(DE-HGF)POF3-344},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:22968170},
pmc = {pmc:PMC3501225},
doi = {10.1038/emboj.2012.261},
url = {https://pub.dzne.de/record/136697},
}