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Opsoclonus myoclonus syndrome (OMS) is a rare neurological disorder characterized by rapid saccadic eye movements, focal myoclonus, and gait disturbances. Both neoplastic and infectious processes have been found to be associated with OMS. In this report, we present a case of OMS in the setting of a new diagnosis of HIV. After diagnosis, the patient was initiated on antiretroviral therapy in addition to clonazepam with some improvement in her neurological symptoms. It was not until she was started on prednisone as an outpatient that her symptoms completely resolved and she returned to her functional baseline.