Hypoglossal neuropathy with associated myokymia as a delayed effect of radiation is a rare occurrence, presumably due to the relative resistance of cranial nerves to injury from irradiation. The authors describe the first case of myokymia of the unilateral tongue with myokymic discharges on needle electromyography after hypofractionated radiation therapy for an extracranial melanoma of the neck. The earlier onset of myokymia than previous cases may represent more direct radiation exposure due to radiation site or the higher radiation dosage administered for treatment of melanomas.