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Obsessive–compulsive disorder (OCD) is characterized by repetitive, persistent and unwanted thoughts and ritualistic, repetitive behaviors. The pathophysiology of OCD involves many distinct cortical and subcortical regions and it has been reported that OCD may occur as a consequence of traumatic brain injury, infections and tumors as well as cerebrovascular insult such as cerebral venous sinus thrombosis (CVST). We here describe the case of a 36-year-old woman who developed OCD at the age of 13 with almost complete remission of the symptoms after a 1 year-long treatment. Interestingly, after suffering CVST at the superior sagittal sinus at the age of 33, she experienced a relapse of OCD. The patient was successfully treated with Sertraline and Clomipramine. Previous studies revealed cases of OCD following different cerebrovascular accidents, i.e. predominantly arterial stroke. However, the present case is the first to describe OCD after venous thrombosis. Based on our clinical experience, the most effective treatment of OCD after CVST represents the combination of the selective serotonin reuptake inhibitor Sertraline and the tricyclic antidepressant Clomipramine.