Recently, accumulating evidence suggests that high bilirubin level is involved in the pathophysiology of schizophrenia. High bilirubin level during early childhood may increase the risk of being suffered from schizophrenia after adulthood, and schizophrenia patients with high bilirubin level have aggravated psychiatric symptoms. As compared with other psychiatric patients and general population, schizophrenia patients usually have relatively higher bilirubin level; high bilirubin level is associated with acute psychotic states, positive symptoms, and poor prognosis in patients with schizophrenia. This article reviews the relation between bilirubin and schizophrenia and its potential pathophysiological mechanism in order to provide a new direction for the study of schizophrenia pathogenesis and auxiliary diagnosis.