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CMT is currently an untreatable disorder. This is partially due to lack of translational models suitable for drug discovery. <jats:italic>In vitro<\/jats:italic> models of CMT have been hindered by the 2D configuration of neuronal cultures, which limits visualization and orientation of axons. To overcome these limitations, we cultured induced pluripotent stem cell (iPSC)\u2010derived spinal motor neurons as 3D spheroids, which grow axons in a centrifugal fashion when plated. Using these iPSC\u2010derived spinal spheroids, we demonstrate neurofilament deposits in motor neuron axons of three patients with CMT2E, caused by mutations in the <jats:italic>NEFL<\/jats:italic> gene. This phenotype is partially reversed by two kinase inhibitors. In summary, we developed a human tridimensional <jats:italic>in vitro<\/jats:italic> system that models length\u2010dependent axonopathies, recapitulates key pathophysiologic features of CMT2E, and should facilitate the identification of new therapeutic compounds for CMT.<\/jats:p>","DOI":"10.1002\/cpt.1718","type":"journal-article","created":{"date-parts":[[2019,11,12]],"date-time":"2019-11-12T18:08:27Z","timestamp":1573582107000},"page":"1231-1239","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":24,"title":["Human Tridimensional Neuronal Cultures for Phenotypic Drug Screening in Inherited Peripheral Neuropathies"],"prefix":"10.1111","volume":"107","author":[{"given":"Renata","family":"Maciel","sequence":"first","affiliation":[{"name":"Department of Neurology University of Miami Miller School of Medicine Miami Florida 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