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@ARTICLE{Waldt:157728,
author = {Waldt, Natalie and Kesseler, Christoph and Fala, Paula and
John, Peter and Kirches, Elmar and Angenstein, Frank and
Mawrin, Christian},
title = {{C}rispr/{C}as-based modeling of {NF}2 loss in meningioma
cells.},
journal = {Journal of neuroscience methods},
volume = {356},
issn = {0165-0270},
address = {Amsterdam [u.a.]},
publisher = {Elsevier Science},
reportid = {DZNE-2021-01185},
pages = {109141},
year = {2021},
abstract = {Alterations of the neurofibromatosis type 2 gene (NF2)
occur in more than fifty percent of sporadic meningiomas.
Meningiomas develop frequently in the setting of the
hereditary tumor syndrome NF2. Investigation of potential
drug-based treatment options has been limited by the lack of
appropriate in vitro and in vivo models.Using Crispr/Cas
gene editing, of the malignant meningioma cell line
IOMM-Lee, we generated a pair of cell clones characterized
by either stable knockout of NF2 and loss of the protein
product merlin or retained merlin protein (transfected
control without gRNA).IOMM-Lee cells lacking NF2 showed
reduced apoptosis and formed bigger colonies compared to
control IOMM-Lee cells. Treatment of non-transfected
IOMM-Lee cells with the focal adhesion kinase (FAK)
inhibitor GSK2256098 resulted in reduced colony sizes.
Orthotopic mouse xenografts showed the formation of
convexity tumors typical for meningiomas with NF2-depleted
and control cells.No orthotopic meningioma models with
genetically-engineered cell pairs are available so far.Our
model based on Crispr/Cas-based gene editing provides paired
meningioma cells suitable to study functional consequences
and therapeutic accessibility of NF2/merlin loss.},
keywords = {Animals / Cell Line, Tumor / Clustered Regularly
Interspaced Short Palindromic Repeats: genetics / Meningeal
Neoplasms: genetics / Meningioma: genetics / Mice /
Neurofibromin 2: genetics / Neurofibromin 2: metabolism /
Crispr/Cas (Other) / Meningioma (Other) / Neurofibromatosis
type 2 (NF2) (Other) / Neurofibromin 2 (NLM Chemicals)},
cin = {AG Angenstein},
ddc = {610},
cid = {I:(DE-2719)1310004},
pnm = {351 - Brain Function (POF4-351)},
pid = {G:(DE-HGF)POF4-351},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:33753124},
doi = {10.1016/j.jneumeth.2021.109141},
url = {https://pub.dzne.de/record/157728},
}