OBJECTIVE: This study aimed to determine the prevalence of autism spectrum disorder (ASD) by using the Autism Diagnostic Observation Schedule-Generic (ADOS-G) classifications in children born very preterm during their toddler years. METHODS: Two birth cohorts of toddlers (2 and 4 years old) each recruited over 12 months and born at <29 weeks' gestation were administered the Modified Checklist of Autism in Toddlers-Follow-up Interview (M-CHAT-FI) screen, the ADOS-G, and developmental assessments. The ADOS-G was conducted on toddlers with M-CHAT-FI-positive screens. RESULTS: Data were available on 88% (169/192) of children. In total, 22 (13%) toddlers screened positive and 3 (1.8%) were confirmed diagnostically with ASD. These 3 cases reached the highest ADOS-G threshold classification of autism. All but 1 child who scored below the ADOS-G thresholds (11/12) demonstrated some difficulty with social communication. Risk was significantly increased for co-occurring neurodevelopmental problems in 21 of the 22 positive-screen ASD cases. Adaptive behavior (P < .001) was the only co-occurring factor independently predictive of ASD in toddlers. CONCLUSIONS: Children born very preterm are at increased risk of ASD. By using the ADOS-G, we found a lower incidence of ASD in children born at <29 weeks' gestation compared with previous studies. Children who screened positive for ASD on the M-CHAT-FI had developmental delays consistent with subthreshold communication impairment.