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Human induced pluripotent stem cells (iPSCs) are expected to represent a useful tool for toxicity testing. However, among many assays, appropriate cellular assays for predicting neurotoxicity in an iPSC\u2010based model are still uncertain. Here we generated neurons from iPSCs of Charcot\u2013Marie\u2013Tooth disease (CMT) patients. Some CMT patients are sensitive to anticancer drugs and present with an adverse reaction of neuropathy. We analyzed cellular phenotypes and found that mitochondria in neurites of CMT neurons were morphologically shorter and showed slower mobility compared to control. A neurosphere assay showed that treatment with drugs known to cause neuropathy caused mitochondrial aggregations in neurites with adenosine triphosphate shortage in both CMT and control neurons, although more severely in CMT. These findings suggest that the genetically susceptible model could provide a useful tool to predict drug\u2010induced neurotoxicity.<\/jats:p>","DOI":"10.1002\/cpt.562","type":"journal-article","created":{"date-parts":[[2016,11,10]],"date-time":"2016-11-10T10:03:48Z","timestamp":1478772228000},"page":"754-762","source":"Crossref","is-referenced-by-count":34,"title":["Modeling Drug\u2010Induced Neuropathy Using Human iPSCs for Predictive Toxicology"],"prefix":"10.1111","volume":"101","author":[{"given":"R","family":"Ohara","sequence":"first","affiliation":[{"name":"Center for iPS Cells for Research and Application (CiRA) Kyoto University Kyoto Japan"},{"name":"Department of Neurology Kyoto Prefectural University of Medicine Kyoto Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"K","family":"Imamura","sequence":"additional","affiliation":[{"name":"Center for iPS Cells for Research and Application (CiRA) Kyoto University Kyoto 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