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• W843516649 abstract "Amyotrophic lateral sclerosis (ALS) is a severe and incurable neurodegenerative disease. Human motor neurons generated from induced pluripotent stem cells (iPSc) offer new perspectives for disease modeling and drug testing in ALS. In standard iPSc-derived cultures, however, the two major phenotypic alterations of ALS--degeneration of motor neuron cell bodies and axons--are often obscured by cell body clustering, extensive axon criss-crossing and presence of unwanted cell types. Here, we succeeded in isolating 100% pure and standardized human motor neurons by a novel FACS double selection based on a p75(NTR) surface epitope and an HB9::RFP lentivirus reporter. The p75(NTR)/HB9::RFP motor neurons survive and grow well without forming clusters or entangled axons, are electrically excitable, contain ALS-relevant motor neuron subtypes and form functional connections with co-cultured myotubes. Importantly, they undergo rapid and massive cell death and axon degeneration in response to mutant SOD1 astrocytes. These data demonstrate the potential of FACS-isolated human iPSc-derived motor neurons for improved disease modeling and drug testing in ALS and related motor neuron diseases." @default.
• W843516649 created "2016-06-24" @default.
• W843516649 creator A5008323486 @default.
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• W843516649 date "2015-10-01" @default.
• W843516649 modified "2023-10-18" @default.
• W843516649 title "Modeling amyotrophic lateral sclerosis in pure human iPSc-derived motor neurons isolated by a novel FACS double selection technique" @default.
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• W843516649 doi "https://doi.org/10.1016/j.nbd.2015.06.011" @default.
• W843516649 hasPubMedId "https://pubmed.ncbi.nlm.nih.gov/26107889" @default.
• W843516649 hasPublicationYear "2015" @default.
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