Disrupted in schizophrenia 1 (DISC1) is one of the strongest supported risk genes for psychiatric disorders, such as schizophrenia, major depression, bipolar disorder, and autism. Intensive study over the past 11 years, since the gene was cloned, has tried to understand at the molecular and cellular levels how mutations in DISC1 contribute to these diseases. The DISC1 protein has been reported to be localized to cytoskeleton-rich regions in cells, including the centrosome, base of primary cilia, axon and dendritic shafts and spines. Here we review the functions of DISC1 which are relevant for cytoskeletal regulation and its crucial roles during normal brain development and in adult brain function. This article is part of a Special Issue entitled Neuronal Function.