The efforts to decipher the genetic causes of schizophrenia, one of the most devastating mental illnesses, have reached a turning point. Several linkage findings in schizophrenia have been replicated and, in the last few years, have been followed by systematic fine-mapping efforts to identify positional susceptibility genes. Here, we outline the evidence supporting each of the proposed positional candidate genes and identify some general areas of caution in their interpretation. Several of these findings hold considerable promise both for understanding the neuropathology of this brain disorder, the causes of which remain a mystery, but also for development of novel, mechanism-based treatments for the patients.