% IMPORTANT: The following is UTF-8 encoded. This means that in the presence
% of non-ASCII characters, it will not work with BibTeX 0.99 or older.
% Instead, you should use an up-to-date BibTeX implementation like “bibtex8” or
% “biber”.
@ARTICLE{Riemenschneider:163937,
author = {Riemenschneider, Henrick and Guo, Qiang and Bader, Jakob
and Frottin, Frédéric and Farny, Daniel and Kleinberger,
Gernot and Haass, Christian and Mann, Matthias and Hartl, F
Ulrich and Baumeister, Wolfgang and Hipp, Mark S and
Meissner, Felix and Fernández-Busnadiego, Rubén and
Edbauer, Dieter},
title = {{G}el-like inclusions of {C}-terminal fragments of {TDP}-43
sequester stalled proteasomes in neurons.},
journal = {EMBO reports},
volume = {23},
number = {6},
issn = {1469-221X},
address = {Hoboken, NJ [u.a.]},
publisher = {Wiley},
reportid = {DZNE-2022-00611},
pages = {e53890},
year = {2022},
note = {(CC BY)},
abstract = {Aggregation of the multifunctional RNA-binding protein
TDP-43 defines large subgroups of amyotrophic lateral
sclerosis and frontotemporal dementia and correlates with
neurodegeneration in both diseases. In disease,
characteristic C-terminal fragments of ~25 kDa ('TDP-25')
accumulate in cytoplasmic inclusions. Here, we analyze
gain-of-function mechanisms of TDP-25 combining
cryo-electron tomography, proteomics, and functional assays.
In neurons, cytoplasmic TDP-25 inclusions are amorphous, and
photobleaching experiments reveal gel-like biophysical
properties that are less dynamic than nuclear TDP-43.
Compared with full-length TDP-43, the TDP-25 interactome is
depleted of low-complexity domain proteins. TDP-25
inclusions are enriched in 26S proteasomes adopting
exclusively substrate-processing conformations, suggesting
that inclusions sequester proteasomes, which are largely
stalled and no longer undergo the cyclic conformational
changes required for proteolytic activity. Reporter assays
confirm that TDP-25 impairs proteostasis, and this
inhibitory function is enhanced by ALS-causing TDP-43
mutations. These findings support a patho-physiological
relevance of proteasome dysfunction in ALS/FTD.},
keywords = {Amyotrophic Lateral Sclerosis: genetics / Amyotrophic
Lateral Sclerosis: metabolism / DNA-Binding Proteins:
genetics / DNA-Binding Proteins: metabolism / Frontotemporal
Dementia: genetics / Frontotemporal Dementia: metabolism /
Humans / Inclusion Bodies: metabolism / Neurons: metabolism
/ Peptide Fragments: genetics / Peptide Fragments:
metabolism / Proteasome Endopeptidase Complex: metabolism /
ALS (Other) / TDP-43 (Other) / neurodegeneration (Other) /
phase separation (Other) / proteasome (Other)},
cin = {AG Edbauer / AG Lichtenthaler / AG Herms / AG Haass},
ddc = {570},
cid = {I:(DE-2719)1110004 / I:(DE-2719)1110006 /
I:(DE-2719)1110001 / I:(DE-2719)1110007},
pnm = {352 - Disease Mechanisms (POF4-352)},
pid = {G:(DE-HGF)POF4-352},
typ = {PUB:(DE-HGF)16},
pmc = {pmc:PMC9171420},
pubmed = {pmid:35438230},
doi = {10.15252/embr.202153890},
url = {https://pub.dzne.de/record/163937},
}