Migraine with aura is a common neurological disorder. Cortical spreading depression is the hypothetical pathophysiologic correlate, but it has never been directly recorded during migraine in a human. A 32-year-old female with a history of migraine underwent a presurgical workup for comorbid epilepsy. While implanted for stereotactic electroencephalography with 94 electrode contacts across bilateral hemispheres, she experienced a typical migraine with aura. Stereotactic electroencephalography demonstrated low-voltage suppression starting in the left mesial occipital cortex and propagating anteriorly at 3 mm/min, clinically correlating with a contralateral right superior scintillating scotoma and ipsilateral headache. Intracranial stereotactic electroencephalographic recording demonstrated cortical spreading depression during migraine with aura. Our findings add to the body of evidence implicating cortical spreading depression in the pathophysiology of migraine with aura.