Background Sexual dysfunction is a common condition in patients taking antipsychotic medication, with a reported prevalence of 45-80% in males, and 30-80% in females (1). Ziprasidone induced spontaneous orgasm (2) and priapism (3) were reported in the literature. Here, we report a case of increased sexual arousal after ziprasidone treatment. Case A twenty-three-year old, single, female patient was admitted to our clinic with reference and persecutory delusions, auditory and visual hallucinations. Her complaints had been persisting for about one month. Her mental status examination revealed the following: attention and concentration were diminished, speech was slow, delusions of reference and other Schneiderian first-rank symptoms, and hallucinations were present, reality testing was impaired, judgment and abstract thinking skills partially preserved. The patient was not able to tolerate her treatment due to side effects. Therefore, it was changed to ziprasidone 40 mg/day and the dosage was increased gradually. After increasing the ziprasidone dose to 60 mg/day, she complained about increased sexual arousal. No other symptoms suggestive of a manic episode were present, and no other medical causes were detected. The patient also said that she never had a similar period in her life previously. Ziprasidone was tapered slowly, and quetiapine 300 mg/day was started. She indicated that increased sexual arousal disappeared after the cessation of ziprasidone. Discussion Ziprasidone has been reported to cause sexual dysfunction at a much lower rate than other antipsychotics. To the best of our knowledge, this is the first case report of increased sexual arousal with ziprasidone.