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@ARTICLE{Arribas:268530,
author = {Arribas, Víctor and Onetti, Yara and Ramiro-Pareta, Marina
and Villacampa, Pilar and Beck, Heike and Alberola, Mariona
and Esteve-Codina, Anna and Merkel, Angelika and Sperandio,
Markus and Martínez-Estrada, Ofelia M and Schmid, Bettina
and Montanez, Eloi},
title = {{E}ndothelial {TDP}-43 controls sprouting angiogenesis and
vascular barrier integrity, and its deletion triggers
neuroinflammation.},
journal = {JCI insight},
volume = {9},
number = {5},
issn = {2379-3708},
address = {Ann Arbor, Michigan},
publisher = {JCI Insight},
reportid = {DZNE-2024-00271},
pages = {e177819},
year = {2024},
abstract = {TAR DNA-binding protein 43 (TDP-43) is a DNA/RNA-binding
protein that regulates gene expression, and its malfunction
in neurons has been causally associated with multiple
neurodegenerative disorders. Although progress has been made
in understanding the functions of TDP-43 in neurons, little
is known about its roles in endothelial cells (ECs),
angiogenesis, and vascular function. Using inducible
EC-specific TDP-43-KO mice, we showed that TDP-43 is
required for sprouting angiogenesis, vascular barrier
integrity, and blood vessel stability. Postnatal EC-specific
deletion of TDP-43 led to retinal hypovascularization due to
defects in vessel sprouting associated with reduced EC
proliferation and migration. In mature blood vessels, loss
of TDP-43 disrupted the blood-brain barrier and triggered
vascular degeneration. These vascular defects were
associated with an inflammatory response in the CNS with
activation of microglia and astrocytes. Mechanistically,
deletion of TDP-43 disrupted the fibronectin matrix around
sprouting vessels and reduced β-catenin signaling in ECs.
Together, our results indicate that TDP-43 is essential for
the formation of a stable and mature vasculature.},
keywords = {Mice / Animals / Endothelial Cells: metabolism /
Neuroinflammatory Diseases / Angiogenesis /
Neovascularization, Physiologic: genetics / DNA-Binding
Proteins: genetics / DNA-Binding Proteins: metabolism /
Angiogenesis (Other) / Endothelial cells (Other) / Vascular
biology (Other) / DNA-Binding Proteins (NLM Chemicals)},
cin = {AG Schmid},
ddc = {610},
cid = {I:(DE-2719)1140002},
pnm = {352 - Disease Mechanisms (POF4-352)},
pid = {G:(DE-HGF)POF4-352},
typ = {PUB:(DE-HGF)16},
pmc = {pmc:PMC11143933},
pubmed = {pmid:38300714},
doi = {10.1172/jci.insight.177819},
url = {https://pub.dzne.de/record/268530},
}