Representative malformations of cortical development, midline anomalies, and hindbrain malformations are discussed with emphasis on radiologic-genetic correlations.<h4>Results</h4>Critical developmental pathways and proteins-including mTOR and Ras/MAPK signaling cascades and the tubulin cytoskeleton-are central to the pathogenesis of congenital brain malformations. The gene discussed is MTOR; the disease is cerebral malformation.