Objective Frontonasal dysplasia is a rare developmental defect of the midface, and little is known about the dental involvement in individuals with this condition. This study investigated tooth abnormalities and occlusal disorders in individuals with frontonasal dysplasia. Design Cross-sectional. Setting Hospital for Rehabilitation of Craniofacial Anomalies, University of São Paulo, Bauru, Brazil. Interventions Clinical oral examination, analysis of patient records, and panoramic radiographs. Participants A total of 20 individuals with frontonasal dysplasia aged 7 to 17 years. Main Outcome Measures Prevalence of the several tooth abnormalities and occlusal disorders analyzed. Results A total of 19 individuals presented at least one tooth abnormality, with highly variable findings. In radiographs, 20% of individuals (all presenting oral clefts) presented agenesis of lateral incisors and second premolars. No supernumerary teeth were observed; 65% of individuals exhibited occlusal alterations, especially anterior open bite in the two individuals with median cleft lip. Conclusions Variable clinical and radiographic alterations were observed, probably due to the large variety of phenotypic characteristics. No specific dental alteration could be related with frontonasal dysplasia.