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- W3092800825 abstract "Nakajo-Nishimura syndrome (NNS) is an autoinflammatory disorder caused by a homozygous mutations in the PSMB8 gene. The administration of systemic corticosteroids is partially effective, but continuous treatment causes severe side effects. We previously established a pluripotent stem cell (PSC)-derived NNS disease model that reproduces several inflammatory phenotypes, including the overproduction of monocyte chemoattractant protein-1 (MCP-1) and interferon gamma-induced protein-10 (IP-10). Here we performed high-throughput compound screening (HTS) using this PSC-derived NNS model to find potential therapeutic candidates and identified CUDC-907 as an effective inhibitor of the release of MCP-1 and IP-10. Short-term treatment of CUDC-907 did not induce cell death within therapeutic concentrations and was also effective on primary patient cells. Further analysis indicated that the inhibitory effect was post-transcriptional. These findings suggest that HTS with PSC-derived disease models is useful for finding drug candidates for autoinflammatory diseases." @default.
- W3092800825 created "2020-10-22" @default.
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- W3092800825 date "2020-10-14" @default.
- W3092800825 modified "2023-09-24" @default.
- W3092800825 title "Pluripotent stem cell‐based screening identifies CUDC ‐907 as an effective compound for restoring the in vitro phenotype of Nakajo‐Nishimura syndrome" @default.
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- W3092800825 doi "https://doi.org/10.1002/sctm.20-0198" @default.
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