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@ARTICLE{Reichelt:599937,
author = {Reichelt, Julia and Sachs, Wiebke and Frömbling, Sarah and
Fehlert, Julia and Studencka-Turski, Maja and Betz, Anna and
Loreth, Desiree and Blume, Lukas and Witt, Susanne and Pohl,
Sandra and Brand, Johannes and Czesla, Maire and Knop, Jan
and Florea, Bogdan I. and Zielinski, Stephanie and Sachs,
Marlies and Hoxha, Elion and Hermans-Borgmeyer, Irm and
Zahner, Gunther and Wiech, Thorsten and Krüger, Elke and
Meyer-Schwesinger, Catherine},
title = {{N}on-functional ubiquitin {C}-terminal hydrolase {L}1
drives podocyte injury through impairing proteasomes in
autoimmune glomerulonephritis},
journal = {Nature Communications},
volume = {14},
number = {1},
issn = {2041-1723},
address = {London},
publisher = {Nature Publishing Group UK},
reportid = {PUBDB-2023-07626},
pages = {2114},
year = {2023},
note = {The authors would like to thank Antonio Virgillio Failla
from the UMIF,UKE and Roland Thuenauer from the ALFM, CSSB,
DESY for technicalassistance in super resolution
microscopy.},
abstract = {Little is known about the mechanistic significance of the
ubiquitin proteasome system (UPS) in a kidney autoimmune
environment. In membranous nephropathy (MN), autoantibodies
target podocytes of the glomerular filter resulting in
proteinuria. Converging biochemical, structural, mouse
pathomechanistic, and clinical information we report that
the deubiquitinase Ubiquitin C-terminal hydrolase L1
(UCH-L1) is induced by oxidative stress in podocytes and is
directly involved in proteasome substrate accumulation.
Mechanistically, this toxic gain-of-function is mediated by
non-functional UCH-L1, which interacts with and thereby
impairs proteasomes. In experimental MN, UCH-L1 becomes
non-functional and MN patients with poor outcome exhibit
autoantibodies with preferential reactivity to
non-functional UCH-L1. Podocyte-specific deletion of UCH-L1
protects from experimental MN, whereas overexpression of
non-functional UCH-L1 impairs podocyte proteostasis and
drives injury in mice. In conclusion, the UPS is
pathomechanistically linked to podocyte disease by aberrant
proteasomal interactions of non-functional UCH-L1.},
cin = {FS-CS / CSSB-CF-PP},
ddc = {500},
cid = {I:(DE-H253)FS-CS-20210408 / I:(DE-H253)CSSB-CF-PP-20210530},
pnm = {633 - Life Sciences – Building Blocks of Life: Structure
and Function (POF4-633)},
pid = {G:(DE-HGF)POF4-633},
experiment = {EXP:(DE-MLZ)NOSPEC-20140101},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:37055432},
UT = {WOS:000978473800001},
doi = {10.1038/s41467-023-37836-8},
url = {https://bib-pubdb1.desy.de/record/599937},
}