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@ARTICLE{Vlkner:165373,
author = {Völkner, Manuela and Wagner, Felix and Steinheuer, Lisa
Maria and Carido, Madalena and Kurth, Thomas and Yazbeck,
Ali and Schor, Jana and Wieneke, Stephanie and Ebner, Lynn
and Del Toro Runzer, Claudia and Taborsky, David and
Zoschke, Katja and Vogt, Marlen and Canzler, Sebastian and
Hermann, Andreas and Khattak, Shahryar and Hackermüller,
Jörg and Karl, Mike Oliver},
title = {{HBEGF}-{TNF} induce a complex outer retinal pathology with
photoreceptor cell extrusion in human organoids.},
journal = {Nature Communications},
volume = {13},
number = {1},
issn = {2041-1723},
address = {[London]},
publisher = {Nature Publishing Group UK},
reportid = {DZNE-2022-01647},
pages = {6183},
year = {2022},
abstract = {Human organoids could facilitate research of complex and
currently incurable neuropathologies, such as age-related
macular degeneration (AMD) which causes blindness. Here, we
establish a human retinal organoid system reproducing
several parameters of the human retina, including some
within the macula, to model a complex combination of
photoreceptor and glial pathologies. We show that combined
application of TNF and HBEGF, factors associated with
neuropathologies, is sufficient to induce photoreceptor
degeneration, glial pathologies, dyslamination, and scar
formation: These develop simultaneously and progressively as
one complex phenotype. Histologic, transcriptome,
live-imaging, and mechanistic studies reveal a previously
unknown pathomechanism: Photoreceptor neurodegeneration via
cell extrusion. This could be relevant for aging, AMD, and
some inherited diseases. Pharmacological inhibitors of the
mechanosensor PIEZO1, MAPK, and actomyosin each avert
pathogenesis; a PIEZO1 activator induces photoreceptor
extrusion. Our model offers mechanistic insights, hypotheses
for neuropathologies, and it could be used to develop
therapies to prevent vision loss or to regenerate the retina
in patients suffering from AMD and other diseases.},
keywords = {Humans / Actomyosin / Heparin-binding EGF-like Growth
Factor / Ion Channels / Macular Degeneration: pathology /
Organoids: pathology / Photoreceptor Cells / Retina:
pathology / Tumor Necrosis Factors / Actomyosin (NLM
Chemicals) / HBEGF protein, human (NLM Chemicals) /
Heparin-binding EGF-like Growth Factor (NLM Chemicals) / Ion
Channels (NLM Chemicals) / PIEZO1 protein, human (NLM
Chemicals) / Tumor Necrosis Factors (NLM Chemicals)},
cin = {AG Karl / Cell culture platform / AG Hermann},
ddc = {500},
cid = {I:(DE-2719)1710004 / I:(DE-2719)1740003 /
I:(DE-2719)1511100},
pnm = {352 - Disease Mechanisms (POF4-352) / 353 - Clinical and
Health Care Research (POF4-353)},
pid = {G:(DE-HGF)POF4-352 / G:(DE-HGF)POF4-353},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:36261438},
pmc = {pmc:PMC9581928},
doi = {10.1038/s41467-022-33848-y},
url = {https://pub.dzne.de/record/165373},
}