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We report a patient with severe orthostatic tremor (OT) unresponsive to pharmacological treatments that was successfully controlled with thalamic (Vim, ventralis intermedius nucleus) deep brain stimulation (DBS) over a 4‐year period. Cortical activity associated with the OT revealed by EEG back‐averaging and fluoro‐deoxi‐glucose PET were also suppressed in parallel with tremor arrest. This case suggests that Vim‐DBS may be a useful therapeutic approach for patients highly disabled by OT. © 2008 Movement Disorder Society