Levetiracetam is an antiepileptic drug with a broad clinical spectrum, effective in focal epilepsy as well as in idiopathic generalized epilepsy. Preclinical animal models and several case reports have found that levetiracetam reduced dystonia. Common adverse effects are asthenia, somnolence, headache and dizziness. We report a case of weight loss associated with the use of levetiracetam. BK is a 21-year-old man, who was presented to a movement disorders clinic of a university hospital for the treatment of primary idiopathic cervical dystonia. Findings of detailed investigations were within normal limits. A medical history and review of his medical records revealed that there was no response to anti-spasticity medications, dopaminergic medications, or benzodiazepines. The patient was treated with levetiracetam in monotherapy, starting with 500 mg daily. After starting levetiracetam, he has lost 8 kg in two months. There was no interference with other medications that are known to have an effect on body weight. Several biological tests were normal. Psychiatric evaluation for weight loss was normal. We identified no other cause of weight loss except using levetiracetam. The patient continued treatment because the body weight trend tended to follow a plateau in the course of long term treatment and his complaints has been reduced. Although levetiracetam is considered to be neutral on body weight, we should keep in mind that levetiracetam could cause weight loss. The mechanisms of weight loss are not exactly known. In our opinion, further data with larger series might provide more information about the weight loss associated with levetiracetam.