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Purpose: Radiation therapy is an independent risk factor for adverse sequelae to the oral cavity and dentition in childhood cancer survivors. However, dental toxicities after radiation therapy often are underreported and there are minimal published data on disturbances in tooth development after proton beam therapy (PBT). We present the long-term clinical and radiographic dental findings 8 years after treatment completion for a patient treated with PBT and chemotherapy for rhabdomyosarcoma. Materials and Methods: Clinical follow-up data of patients treated with PBT within the Proton Overseas Programme (POP) is stored in a National Database and curated by a dedicated outcomes unit at the Christie NHS PBT center. This case report was identified from the extraction and analysis of data for pediatric head and neck cancer patients in this database for a service evaluation project. Results: The permanent dentition in this patient aged 3.5 years at the time of treatment was severely affected with abnormal dental development first observed 3.5 years after treatment completion. PBT delivered mean doses of 30 Gy(RBE = 1.1) to the maxilla and 25.9 Gy(RBE = 1.1) to the mandible. Conclusion: Significant dental development abnormalities occurred in this pediatric patient, despite doses in areas being lower than the proposed thresholds in the literature. Improved descriptions of dental toxicities and routine contouring of the maxilla and mandible are needed to correlate dosimetric data. The dose to teeth should be kept as low as reasonably possible in younger patients until the dose thresholds for dental toxicities are known.