Background: The cerebellum has traditionally been regarded as an organ of motor coordination. However, the importance of the cerebellum in psychiatric disorders, behavior, and cognition is increasingly being recognized. There is no consensus concerning treatment of schizophrenia-like psychosis after cerebellar pathology. Reports describe the use of several antipsychotics, either alone or in combination with antidepressants or lithium. Clozapine is used for the treatment of refractory schizophrenia, but there are no reports of its use in the abovementioned situation.Case Presentation: We report the case of a 20-year-old woman who developed a schizophrenia-like psychosis after rupture of arteriovenous malformation at 13 years of age. The psychotic symptoms proved to be resistant to treatment, and several psychopharmacological schemas were tried. The clinical picture only showed consistent improvement with the combined use of clozapine and valproate. Conclusions:The relationship between chronic psychosis and cerebellar pathology remains poorly understood. Cases like the present one suggest that clozapine and valproate may be used safely and effectively in refractory schizophrenia-like psychosis associated with cerebellar pathology.