Journal of Maxillofacial and Oral Surgery, 2026 (ESCI, Scopus)
Introduction: The calcifying odontogenic cyst is a rare and generally benign odontogenic epithelial lesion. Reports of its occurrence in the pediatric population and within the maxillary sinus are particularly scarce. In this study, we present a rare case of this entity associated with a compound odontoma, which was successfully managed using an endoscope-assisted surgical approach. This case represents a unique contribution to the literature in terms of both its unusual localization and the innovative surgical technique employed. Case Report: We report a case of a calcifying odontogenic cyst in an 11-year-old male patient who presented with painless swelling in the left maxilla. Clinical and radiographic evaluations revealed a large, unilocular, calcified lesion containing multiple tooth-like structures, extending toward the maxillary sinus and the infraorbital rim. The lesion was completely enucleated under general anesthesia, and the associated odontoma-like structures and supernumerary teeth were excised. Histopathological analysis confirmed the diagnosis of a calcifying odontogenic cyst associated with a compound odontoma. Conclusion: Although the coexistence of a calcifying odontogenic cyst and a compound odontoma is rare, it should be considered in the differential diagnosis of painless maxillary swellings in pediatric patients. These lesions, which exhibit characteristic radiographic features, should be thoroughly evaluated using advanced imaging modalities and managed by complete surgical excision. No recurrence was observed during a 24-month follow-up period; however, long-term clinical and radiological monitoring is recommended due to the low risk of recurrence and the potential for malignant transformation.