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Priapism, an uncommon urological emergency, is a pathologically prolonged and painful penile erection, usually unassociated with sexual desire or intercourse. Drug-induced priapism comprises about 30% of the cases and it’s estimated that 50% of them were induced by antipsychotic agents Although typical antipsychotics are often associated with priapism, there are some case reports which reported clozapine, risperidone, olanzapine, quetiapine and aripiprazole as a cause of priapism. There is no case report about priapism with only amisulpride usage. In this article we present a case of priapism caused by the use of amisulpride. A 26-year-old, primary school graduate, single and unemployed male. The patient was brought to the clinic by his family with disorganized speech and behavior, and persecutory delusions. Patient’s complaints started 5 years ago. The first diagnosis was made as “schizophrenia”; treatment is arranged as a combination of şupentixol depot (1 injection per 15 days) and olanzapine 20 mg/day. Patient’s complaints are partially reduced after using these drugs on a regular basis. Because of weight gain, treatment changed with risperidone and chlorpromazine. Because of increase in patient’s complaints with this treatment, the patient’s family brought the patient to our clinic. According to medical history taken from the patient and their relatives, there was no history of substance abuse. To arrange treatment, patient was admitted to our service. The patient’s physical and neurological examinations and blood tests were normal. The patient was diagnosed to have schizophrenia according to DSM-IV. Increasing the dose of Amisulpride by 200 mg per week, a dose of 800mg/day is attained. In the first day of using 800 mg amisulpride, involuntary, painful erection complaint was observed that lasted about 6 hours. As a result of a joint consultation with the Department of Urology, priapism was diagnosed. There was no physical disease and perineal trauma which may cause priapism. So that was thought to be related with the use of amisulpride and amisulpiride treatment was stopped. Patient had been followed up for 3 days without medication and during this period priapism wasn’t observed. A literature search based on the key words “priapism”, “amisulpride” and “atypical antipsychotics” yields no case report about priapism that was associated to sole use of amisulpride. This case is interesting because it is the first case associated with priapism caused by amisulpride. Priapism, may cause erectile dysfunction through penile tissue ischemia; for this reason diagnosis and treatment is important. Priapism may occur at any stage of antipsychotic medication and is not possible to predict. On patients treated with psychotropic drugs, erections which lasted between 15-20 minutes, painless and long-lasting without any sexual activity, could be a sign of priapism. In these cases, stopping the patient’s medication, may be sufficient to prevent the development of priapism.