Wilson's disease consists in a inborn error of metabolism with marked neuropsychiatric manifestations. These manifestations need to be correctly identified and treated. There are four neuropsychiatric symptoms clusters established for Wilson's disease patients: behavior and/or personality disorders, mood disorders, cognitive deficits, and psychotic manifestations. Although such patients are more commonly seen in neurological and hepatological settings, mental health professionals must keep in mind a high level of suspicion, once first presentations may be of psychiatric nature. This study presents a review of literature about neuropsychiatric aspects of Wilson's disease, emphasizing diagnostic issues. It is presented the case of a 26 years old male patient initially with psychiatric polimorfic symptoms. The treatment of this patient with electroconvulsive therapy is presented, adding a report of this therapeutic modality, applied to that case, to the two only previous records in the literature.61876-80