Children with genetic syndromes offer a unique opportunity to combine genetic and environmental approaches to the study of aggression. Children with genetic syndromes associated with developmental delay are at increased risk for behavior problems, but little is known about risk and resilience factors. In this study, we examined maternal sensitivity of mothers of children with Down syndrome using home observations when their children were 2, 3, and 5 years old, and relations with maternal reports and observations of overt aggression at school at age 5. Maternal sensitivity at ages 2 and 3 years did not significantly predict child aggression at age 5, but low maternal sensitivity at age 5 was significantly related to overt aggression at both home and school. By replicating and extending earlier work, this study informs developmental theory and identifies an important maternal variable related to aggression in children with Down syndrome. (Contains 1 table.)