The present study illustrates a case of obsessive-compulsive disorder (OCD) with schizotypal personality treated by social skills training. Prior research suggests that OCD with schizotypal personality predicts poor treatment outcome using exposure-based treatments. Following social skills treatment and at 6-month follow-up, the patient had considerable obsessive-compulsive symptom reduction, although he was still symptomatic for OCD, anxiety and depression. Controlled trails are indicated to illuminate the specific contributions of this approach for OCD with schizotypal personality disorder.