Dystonia, which is characterized by involuntary and uncontrolled muscle contractions, is caused by treatment of typical antipsychotic drugs in psychiatric practices. Although various antidepressants associated with dystonia were reported in literature; dystonia resulting from bupropion treatment are limited. A 34-year-old female, the patient with no history of a psychiatric or medical disorder, was prescribed şuoxetine 20-40 mg/day. In a two-months period of treatment, because of lack of response, şuoxetine 20 mg/day was augmented with bupropion XL (extended-release) 150 mg/day. On the follow-up two weeks later, şuoxetine 20 mg/day was discontinued and the dosage of bupropion XL (extended-release) was increased to 300 mg/day. In the third week of bupropion XL (extended-release) 300 mg/day treatment, she developed jaw clenching, inability to rotate her head, limited tongue movements and swallowing difficulty. There were no add-on medications and blood cell count and all biochemical parameters including electrolytes, renal and hepatic function tests and thyroid function tests were within normal range. On third day of reduction of the bupropion dosage from 300 mg/day to 150 mg/day, symptoms had decreased and three days of follow up after termination of bupropion treatment revealed complete regression of symptoms. There was no medical disorder in addition to depression or no medication in addition to bupropion. Three weeks of follow up with titration of bupropion dosage from 150 mg/day to 300 mg/day led to dystonia symptoms and complete remission of symptoms was observed with reduction-termination of bupropion treatment. This case suggests that physicians should be careful about dystonia as a side-effect associated with bupropion XL (extended-release) in clinical practice.