The advent of the genomic era has led to the discovery of linkages of several genes and pathways to schizophrenia and autism spectrum disorder (ASD) that may serve as new biomarkers or therapeutic targets for these diseases. Two large-scale genetic studies published early in 2011 provided evidence that functional microduplications at 7q36.3, containing VIPR2, are a risk factor for schizophrenia. 7q36.3 microduplications were also reported to be significantly increased in ASD. VIPR2 encodes VPAC2, a seven transmembrane heterotrimeric G protein-coupled receptor that binds two homologous neuropeptides with high affinity, PACAP and VIP. These clinical studies demonstrate a VIPR2 genetic linkage to schizophrenia and ASD and should lead to novel insights into the etiology of these mental health disorders. However, the mechanism by which overactive VPAC2 signaling may lead to schizophrenia and ASD is unknown. In the present review, we will describe recent advances in the genetics of schizophrenia and attempt to discuss the pathophysiological role of altered VPAC2 signaling in psychiatric disorders.