Orthostatic tremor as possible presenting sign of progressive supranuclear palsy

•Orthostatic tremor plus syndromes can be a presenting sign of movement disorders. We suggest remaining vigilant for other extrapyramidal symptoms during OT follow-up.•Orthostatic tremor is often difficult to treat but can sometimes respond to bilateral thalamic deep brain stimulation (DBS).•Diagnosis of PSP can be a challenge during the first years and MRI changes often only become apparent in later stages.