Multimodal cancer therapy for pediatric head and neck tumors may be associated with significant developmental orofacial morbidity. This report details these effects in a child (C.I.) diagnosed at 2.5 years of age with a rhabdomyosarcoma, primary to the left buccinator. This case is of interest as C.I. has an unaffected identical twin (D.I.) for comparative study. Both were assessed by comparing panoramic radiographs and lateral and frontal tracings of cephalometric radiographs obtained at 8.25 years of age. C.I. had multiple dental anomalies which included agenesis, ectopia, crown malformation, and root malformation. Root malformation, ectopia, and agenesis were restricted to the left dentition, whereas crown malformation was noted bilaterally. C.I. had a generalized craniofacial skeletal hypoplasia relative to D.I. in all three planes, growth defects were greater on the side of the tumor, and the mandible was affected more than the nasomaxillary complex.