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Chemical Screening Identifies Enhancers of Mutant Oligodendrocyte Survival and Unmasks a Distinct Pathological Phase in Pelizaeus-Merzbacher Disease.

Stem cell reports - 11 Sept 2018

Elitt Matthew S, Shick H Elizabeth, Madhavan Mayur, Allan Kevin C, Clayton Benjamin L L, Weng Chen, Miller Tyler E, Factor Daniel C, Barbar Lilianne, Nawash Baraa S, Nevin Zachary S, Lager Angela M, Li Yan, Jin Fulai, Adams Drew J, Tesar Paul J

Abstract excerpt

Pelizaeus-Merzbacher disease (PMD) is a fatal X-linked disorder caused by loss of myelinating oligodendrocytes and consequent hypomyelination. The underlying cellular and molecular dysfunctions are not fully defined, but therapeutic enhancement of oligodendrocyte survival could restore functional myelination in patients. Here we generated pure, scalable quantities of induced pluripotent stem cell-derived...

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Identifiers

PMID: 30146490
DOI: 10.1016/j.stemcr.2018.07.015
PMCID: PMC6135742

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