Highlights • Bona fide oculomotor neurons can be derived from stem cells by PHOX2A overexpression • In vitro- and in vivo-generated oculomotor neurons are transcriptionally similar • Stem cell-derived oculomotor neurons display a robust activation of Akt signaling • In vitro-generated oculomotor neurons are relatively resilient to ALS-like toxicity Summary Oculomotor neurons, which regulate eye movement, are resilient to degeneration in the lethal motor neuron disease amyotrophic lateral...