Increased rates of clinically significant internalizing disorders (obsessive compulsive disorder, anxiety, and depression) have been demonstrated in males with Duchenne muscular dystrophy, and a Duchenne muscular dystrophy neuropsychiatric syndrome has been suggested. Although symptoms of depression are widely recognized, some of the other internalizing symptoms are less frequently identified. Through a retrospective chart review of 107 males with Duchenne muscular dystrophy, we identified 15 patients with obsessive compulsive disorder spectrum symptoms; 11 of those also had anxiety symptoms. Many of these patients received selective serotonin reuptake inhibitor treatment, commonly noting improvement in symptoms. Here we describe the clinical features of several patients in detail to facilitate early recognition and consideration for treatment for patients with Duchenne muscular dystrophy and internalizing psychiatric symptoms. The results of this cohort showed a significantly increased rate of obsessive compulsive disorder spectrum symptoms (14%) compared to the general population.