Orthostatic tremor (OT) is a rare movement disorder that consists of involuntary shaking of the legs and trunk present only on standing. Although the origin and the mechanism of this condition are not well understood, the neurophysiologic abnormalities and PET studies suggest a central origin. We describe the clinical and radiologic features of two patients with symptomatic OT and associated pontine lesions, and conclude that OT may arise from dysfunction of the cerebellum or related pontine structures.