Updated findings from the ongoing National Institute of Mental Health Child Psychiatry Branch longitudinal study of childhood-onset schizophrenia (COS) are presented, along with replications from collaborators at other sites. Clinical and neurobiologic continuities of COS with poor-outcome adult-onset schizophrenia suggests that their underlying pathophysiology is the same. However, these early-onset cases appear to have more striking genetic contributions to their etiology. Updated findings involve risk factors (birth complications, cytogenic abnormalities, early language and motor problems, and familial psychopathology), treatment trials, and brain magnetic resonance imaging studies.