TY - JOUR AU - Waldt, Natalie AU - Kesseler, Christoph AU - Fala, Paula AU - John, Peter AU - Kirches, Elmar AU - Angenstein, Frank AU - Mawrin, Christian TI - Crispr/Cas-based modeling of NF2 loss in meningioma cells. JO - Journal of neuroscience methods VL - 356 SN - 0165-0270 CY - Amsterdam [u.a.] PB - Elsevier Science M1 - DZNE-2021-01185 SP - 109141 PY - 2021 AB - 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. KW - Animals KW - Cell Line, Tumor KW - Clustered Regularly Interspaced Short Palindromic Repeats: genetics KW - Meningeal Neoplasms: genetics KW - Meningioma: genetics KW - Mice KW - Neurofibromin 2: genetics KW - Neurofibromin 2: metabolism KW - Crispr/Cas (Other) KW - Meningioma (Other) KW - Neurofibromatosis type 2 (NF2) (Other) KW - Neurofibromin 2 (NLM Chemicals) LB - PUB:(DE-HGF)16 C6 - pmid:33753124 DO - DOI:10.1016/j.jneumeth.2021.109141 UR - https://pub.dzne.de/record/157728 ER -