Tardive dyskinesia is a movement disorder that develops during the course of long-term treatment with neuroleptic agents and is characterized primarily by choreiform and athetotic movements. We report the case of a 68-year old female suffering from Bipolar disorder, treated with lithium monotherapy 600 mg per day (serum levels 0.6) for the last 15 years. While her response was favorable she was rarely visiting the outpatient clinic. Over the last few months she developed a tardive dyskinesia-like syndrome. To our knowledge this is the first reported case of tardive dyskinesia with lithium monotherapy at low dosages.