Although the inheritable nature of schizophrenia is well-established, the genetic underpinnings remain largely hidden. Recently, two independent research groups identified microdeletions conferring high risk of schizophrenia. The deletions are recurrent in nature and offer an explanation to the apparently stable prevalence of the disease, despite reduced fecundity in patients. The findings may lead to development of diagnostic tools and construction of new disease models to help the development of novel therapeutic strategies.