In this, the largest schizophrenia imaging-genetic study conducted to date, we provide the first longitudinal evidence to link a genetic variant impacting DA signaling to structural cortical deficits in children with schizophrenia as well as in their non-psychotic siblings. This suggests that disruption of the causal pathways linking variation in DA signaling to cortical maturation may partly index primary risk factors for schizophrenia rather than solely resulting from the presence of active disease.