Ocular neuromyotonia is a rare disorder of the oculomotor nerves caused by chronic damage to the nerve, usually secondary to previous irradiation or longstanding compression. We present the case of a 40-year-old man who had received proton beam therapyfor a right cavernous sinus chondrosarcoma 15 years earlier. He developed intermittent episodes of self-limited horizontal diplopia, which occurred over the course of 1 year. At its peak, the deviation reached 20Δ in primary gaze and 25Δ in right gaze. Signs included right upper lid retraction and right medial rectus muscle absence of relaxation after sustained left gaze, with mild deficit of elevation and depression but no pupillary involvement. Full recovery occurred after he closed his eyes. Ocular neuromyotonia of the right oculomotor nerve was diagnosed after recurrence of primary disease relapse was ruled out. Symptoms remitted completely under oxcarbazepine.