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@ARTICLE{LlorensTorres:139239,
author = {Llorens Torres, Francesc Josep and Thüne, Katrin and
Sikorska, Beata and Schmitz, Matthias and Tahir, Waqas and
Fernández-Borges, Natalia and Cramm, Maria and Gotzmann,
Nadine and Carmona, Margarita and Streichenberger, Nathalie
and Michel, Uwe and Zafar, Saima and Schütz, Anna-Lena and
Rajput, Ashish and Andréoletti, Olivier and Bonn, Stefan
and Fischer, Andre and Liberski, Pawel P and Torres, Juan
Maria and Ferrer, Isidre and Zerr, Inga},
title = {{A}ltered {C}a2+ homeostasis induces {C}alpain-{C}athepsin
axis activation in sporadic {C}reutzfeldt-{J}akob disease.},
journal = {Acta Neuropathologica Communications},
volume = {5},
number = {1},
issn = {2051-5960},
address = {London},
publisher = {Biomed Central},
reportid = {DZNE-2020-05561},
pages = {35},
year = {2017},
abstract = {Sporadic Creutzfeldt-Jakob disease (sCJD) is the most
prevalent form of human prion disease and it is
characterized by the presence of neuronal loss, spongiform
degeneration, chronic inflammation and the accumulation of
misfolded and pathogenic prion protein (PrPSc). The
molecular mechanisms underlying these alterations are
largely unknown, but the presence of intracellular neuronal
calcium (Ca2+) overload, a general feature in models of
prion diseases, is suggested to play a key role in prion
pathogenesis.Here we describe the presence of massive
regulation of Ca2+ responsive genes in sCJD brain tissue,
accompanied by two Ca2+-dependent processes: endoplasmic
reticulum stress and the activation of the cysteine
proteases Calpains 1/2. Pathogenic Calpain proteins
activation in sCJD is linked to the cleavage of their
cellular substrates, impaired autophagy and lysosomal
damage, which is partially reversed by Calpain inhibition in
a cellular prion model. Additionally, Calpain 1 treatment
enhances seeding activity of PrPSc in a prion conversion
assay. Neuronal lysosomal impairment caused by Calpain over
activation leads to the release of the lysosomal protease
Cathepsin S that in sCJD mainly localises in axons, although
massive Cathepsin S overexpression is detected in microglial
cells. Alterations in Ca2+ homeostasis and activation of
Calpain-Cathepsin axis already occur at pre-clinical stages
of the disease as detected in a humanized sCJD mouse
model.Altogether our work indicates that unbalanced
Calpain-Cathepsin activation is a relevant contributor to
the pathogenesis of sCJD at multiple molecular levels and a
potential target for therapeutic intervention.},
keywords = {Animals / Brain: metabolism / Brain: pathology / Calcium:
metabolism / Calpain: metabolism / Cathepsins: metabolism /
Cations, Divalent: metabolism / Cells, Cultured /
Creutzfeldt-Jakob Syndrome: metabolism / Creutzfeldt-Jakob
Syndrome: pathology / Disease Models, Animal / Homeostasis:
physiology / Humans / Lysosomes: metabolism / Lysosomes:
pathology / Mesocricetus / Mice, Transgenic / Neurons:
metabolism / Neurons: pathology / PrPSc Proteins: metabolism
/ Rats, Wistar / Recombinant Proteins: metabolism / Sheep /
Cations, Divalent (NLM Chemicals) / PrPSc Proteins (NLM
Chemicals) / Recombinant Proteins (NLM Chemicals) /
Cathepsins (NLM Chemicals) / Calpain (NLM Chemicals) /
Calcium (NLM Chemicals)},
cin = {AG Zerr / Ext UMG Zerr / AG Bonn 2 / AG Bonn 1 / AG
Fischer},
ddc = {610},
cid = {I:(DE-2719)1440011-1 / I:(DE-2719)5000037 /
I:(DE-2719)1440012 / I:(DE-2719)1410003 /
I:(DE-2719)1410002},
pnm = {342 - Disease Mechanisms and Model Systems (POF3-342) / 344
- Clinical and Health Care Research (POF3-344)},
pid = {G:(DE-HGF)POF3-342 / G:(DE-HGF)POF3-344},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:28449707},
pmc = {pmc:PMC5408381},
doi = {10.1186/s40478-017-0431-y},
url = {https://pub.dzne.de/record/139239},
}