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@PHDTHESIS{Offermann:258582,
author = {Offermann, Nina},
title = {{R}ole of the proton channel {HVCN}1 in the regulation of
neutrophil activation},
school = {Rheinische Friedrich-Wilhelms-Universität Bonn},
type = {Dissertation},
reportid = {DZNE-2023-00611},
pages = {126},
year = {2023},
note = {Dissertation, Rheinische Friedrich-Wilhelms-Universität
Bonn, 2023},
abstract = {Excessive neutrophil activation is a feature of many
inflammatory diseases. Neutrophils from affected patients
are prone to release reactive oxygen species (ROS) and toxic
granule proteins or to undergo NETosis, a special form of
cell death characterized by the release of extracellular DNA
traps (NETs). Indeed, neutrophils have been shown to play a
critical role in the development of autoimmune diseases, as
NETs provide autoantigens, such as double-stranded DNA in
systemic lupus erythematosus (SLE) or myeloperoxidase (MPO)
in ANCA-associated vasculitis. Nonetheless, the molecular
switches regulating neutrophil effector functions remain to
be further characterized.HVCN1 is the only voltage-gated
proton channel in mammals that regulates pH and membrane
potential during neutrophil activation. Its function
supports the electrogenic activity of the ROS-generating
enzyme NADPH oxidase. Therefore, NADPH oxidase-dependent ROS
production is significantly impaired in the absence of
HVCN1. However, not much is known about how HVCN1 regulates
neutrophil downstream functions.Therefore, in the first part
of my thesis, I further investigated the role of HVCN1 in
neutrophils. I demonstrated that PMA-stimulated NETosis and
MPO release were significantly increased in HVCN1−/−
neutrophils. Fittingly, HVCN1−/− neutrophils showed
atypical mobilization of calcium, increased histone
citrullination, and high mitochondrial ROS caused by
calcium-dependent mitochondrial depolarization. Increased
NETosis, MPO release, and mitochondria ROS could be rescued
by intracellular calcium chelation but not by antioxidants,
suggesting that calcium is the primary determinant of
increased NETosis susceptibility in HVCN1−/−
neutrophils.I further examined the consequences of HVCN1
deficiency in vivo. In agreement with previous reports,
young HVCN1−/− mice did not exhibit a pathogenic
phenotype, however we detected a mild (auto-) inflammatory
phenotype in aged, female HVCN1−/− mice. Likewise,
HVCN1−/− mice showed more severe pathology in two models
of inflammation. Correspondingly, we found HVCN1 to be
downregulated in neutrophils from SLE patients in two
separate datasets. This is the first study to show that
HVCN1 deficiency increases the inflammatory capacity of
murine neutrophils in vitro and in vivo, suggesting that
downregulation of HVCN1 may have implications for human
disease.},
cin = {AG Capasso / Library and Information Services},
cid = {I:(DE-2719)1013033 / I:(DE-2719)1040260},
pnm = {352 - Disease Mechanisms (POF4-352)},
pid = {G:(DE-HGF)POF4-352},
typ = {PUB:(DE-HGF)11},
urn = {urn:nbn:de:hbz:5-71080},
url = {https://pub.dzne.de/record/258582},
}