Opsoclonus is a bizarre oculomotor disturbance consisting of rapid, involuntary, repetitive conjugate eye movements in all directions. Clinically, it has been associated with infections, neoplasms, toxins, and drugs. Pathologically, there is no apparent consistently defined structural lesion. A case of opsoclonus with postmortem results is reported. Opsoclonus in adults and available autopsy data are reviewed.