Aspergeras syndrome (AS) is under-recognized and may be misdiagnosed as schizophrenia in adults because of symptom overlap. Pharmacological treatment usually targets associated behavioral and mental symptoms rather than the actual core features of AS. We report a middle-aged male patient who, after many years of previous contact with mental health services, and on account of his psychotic symptoms and diagnosis of schizophrenia, was admitted to an inner-city acute psychiatric unit, where a primary diagnosis of AS was established for the first time in his life. His impairing clinical features of AS improved markedly following treatment using aripiprazole, a novel atypical antipsychotic that acts as a partial agonist at dopamine D sub(2) receptors. As well as sharing clinical features, there is an overlap in underlying neurobiology of AS and schizophrenia, including dopamine dysfunction, that provides a rationale for using antipsychotics of this class in the clinical management not only of associated psychotic symptoms but also of the core features of AS itself.