We treated a 34-year-old male patient with a diagnosis of both schizophrenia and Brugada syndrome (BrS). In the literature we found clues for ion channel defects as a possible common pathophysiological mechanism in these disorders. Awareness of the link with BrS can improve cardiac assessment and cardiac mortality in patients with schizophrenia, whereas the ion channel defects provide an interesting area for future research into pharmacotherapeutic possibilities.