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Human sexuality is composed of a complex interaction of biological and psychological conditions. Orgasm is a sudden discharge of accumulated sexual tension during the sexual response cycle, resulting in rhythmic muscular contractions in pelvic region characterized by sexual pleasure. Persistent Genital Arousal Disorder (PGAD) is a spontaneous, persistent, and uncontrollable genital arousal in women, with or without orgasm or genital engorgement, unrelated to any feelings of sexual desire. PGAD, first defined in modern literature in 2001, is a rare condition. Women with PAGD report a higher degree of psychological distress and even suicidal thoughts. Probable causes of PGAD are still controversial. Besides psychological stress factors, genital nerve sensitivity, intracranial abnormalities, vascular abnormalities of genitalia and some medications are still considered as possible causes of PGAD. Herein, we present a case that was treated with an antidepressant and had PGAD after cessation of antidepressant. A 39-year-old female patient applied to emergency room with persistent genital arousal symptoms. She had been using duloxetine 60 mg/day for treatment of her depressive symptoms and stopped duloxetine due to its side effects 2 days ago. She had hypothyroidism and she was on thyroid replacement medication. 5 years ago, she used antidepressant treatment for one year. She had no other medical problem in her knowledge. With PGAD pre-diagnosis, all tests are completed to find out the etiology. In this process, her involuntary genital arousals continued to be observed. No significant organic pathology was detected and duloxetine treatment was restarted. With restarting of medication, her complaints were disappeared. PGAD is a rare condition in women that causing a lot of suffering. The proposed etiologies of PGAD are multiple and may involve a range of psychological, pharmacological, neurological and vascular cases. In our case, no factor was found in investigation for finding out the etiology of PGAD. Because of the recovery of the PGAD after restarting duloxetine medication to the patient, it can be considered that, this case was connected with drug-withdrawal. Discontinuing selective serotonin reuptake inhibitor (SSRI) medication suddenly may lead to SSRI Discontinuation Syndrome and its effects may continue more than 18 months. SSRI Discontinuation Syndrome is one of the possible causes of PGAD. In this case, it is thought that PGAD had occurred with the serotonergic mechanism. Although it is not one of the main causes of PGAD; it is important to consider that PGAD may arise due to antidepressant withdrawal.