The syndrome of mania or hypomania (mood elevation with psychomotor agitation, distractibility, pressure of speech, inflated self-esteem, and impulsivity) may arise following deep brain stimulation (DBS) of the subthalamic nucleus (STN) for Parkinson’s disease (PD). 1, 2 Previous reports have attributed the onset of mood changes to microlesional effects during neurosurgery, 3 the amplitude (voltage) of stimulation, 4 or the position of the active electrode contact within the STN. 5 Remediation of mania has been described through reducing the amplitude of stimulation, 4 moving the active contact, 5, 6 delivering a more focused stimulation field by using the electrode as the anode, 6 or the addition of mood-stabilizing medication. 7 Here we report a case of mania with psychotic symptoms in a patient without a personal or family history of bipolar disorder, with persistence after prolonged cessation of stimulation (6 weeks). To our knowledge this is the first such case reported in the literature. This case also describes the phenomenon of a sustained mood change that was positively evaluated by the patient and his family, with emergence of clinically significant symptoms only after activation of an additional electrode contact 9 months after device implantation.