Sphingolipids are biologically active lipids ubiquitously expressed in all vertebrate cells, especially those in the CNS. Aside from their essential roles as structural components of cell membranes, studies over the past two decades have shown that they play vital roles in cellular signaling, cell differentiation and proliferation, apoptosis and inflammation. Given these properties, it is not surprising that disruption of sphingolipid metabolism is strongly associated with several diseases that exhibit diverse neurological, psychiatric, and metabolic consequences. Here, we review the emerging roles of sphingolipids in disease pathogenesis in psychiatric disorders, including schizophrenia, bipolar disorder and major depression. Understanding sphingolipid metabolism and it dysregulation in human disease is significant for the development of new therapeutic approaches.