A case is reported of chronic otitis media and cholesteatoma in an adult patient with behavioral disorders who developed a pyogenic brain abscess. Initially, clinical management focused on the otogenic disease in view of the patient's symptoms and medical history. Finally, less easily recognized discriminating signs and symptoms, and the results of imaging studies were emphasized. These data confirmed the clinical diagnosis of pyogenic brain abscess secondary to cholesteatoma. The case illustrates how one of the most severe infective complications related with cholesteatoma may appear and its a remainder to clinicians to take this possibility into consideration.