Abstract
Odontogenic keratocyst is a benign odontogenic lesion characterized by locally aggressive behavior and a tendency to recur. Its occurrence in the maxilla with maxillary sinus involvement is uncommon and may pose diagnostic and therapeutic challenges because of its proximity to relevant anatomical structures. We report the case of a 22-year-old male patient who presented with swelling in the right maxilla, occasional epiphora, and intermittent rhinorrhea. Panoramic radiography and computed tomography revealed an extensive unilocular radiolucent lesion in the right maxilla, with maxillary sinus involvement, cortical thinning, deviation of the nasal cavity, and extension toward adjacent anatomical regions. Aspiration and incisional biopsy supported the diagnosis, which was subsequently confirmed by histopathological examination as a parakeratinized odontogenic keratocyst. Management consisted of surgical enucleation, cavity debridement, and adjunctive topical application of 5-fluorouracil using sterile gauze maintained in situ for 24 hours. Clinical and imaging follow-up at one week, one month, three months, and six months showed favorable healing, partial bone regeneration, and no clinical or tomographic evidence of early recurrence. This case highlights the value of comprehensive clinical and radiological assessment and suggests that enucleation combined with topical 5-fluorouracil may be a feasible adjunctive alternative in selected anatomically complex maxillary odontogenic keratocysts. However, given the risk of late recurrence, long-term follow-up remains essential.
References
1. Soluk-Tekkesin M, Wright JM. The World Health Organization Classification of Odontogenic Lesions: A Summary of the Changes of the 2022 (5th) Edition. Turk Patoloji Derg. 2022;38(2):168-184. doi:10.5146/tjpath.2022.01573.
2. Vered M, Wright JM. Update from the 5th Edition of the World Health Organization Classification of Head and Neck Tumors: Odontogenic and Maxillofacial Bone Tumours. Head Neck Pathol. 2022;16(1):63-75. doi:10.1007/s12105-021-01404-7.
3. Johnson NR, Gannon OM, Savage NW, Batstone MD. Frequency of odontogenic cysts and tumors: a systematic review. J Investig Clin Dent. 2014;5(1):9-14. doi:10.1111/jicd.12044.
4. Walsh M, Hussein MA, Carter M, Abdulrahman S. Maxillary odontogenic keratocyst. J Surg Case Rep. 2022;2022(4):rjac078. doi:10.1093/jscr/rjac078.
5. Singh S, Shukla P, Bedi RS, Gupta S, Acharya S. An unusual case of maxillary sinus odontogenic keratocyst: an insightful report with review of the literature. Cureus. 2023;15(4):e37357. doi:10.7759/cureus.37357.
6. Zou B, Ding SX, Ru L, An FX, Li YG. Odontogenic keratocyst in maxillary sinus with ectopic third molar: a case report. Am J Case Rep. 2024;25:e944543. doi:10.12659/AJCR.944543.
7. Maruthamuthu K, Vasupradha G, Dineshshankar J, Balaji AR. Recurrent keratocystic odontogenic tumor of right maxillary sinus involving the right infraorbital rim. Natl J Maxillofac Surg. 2017;8(1):70-74. doi:10.4103/0975-5950.208974.
8. Jeon WY, Park JH, Ku JK, Baek JA, Ko SO. Is conservative treatment (enucleation using modified Carnoy’s solution) of odontogenic keratocyst in the maxilla good prognosis? J Korean Assoc Oral Maxillofac Surg. 2023;49(5):287-291. doi:10.5125/jkaoms.2023.49.5.287.
9. Ledderhof NJ, Caminiti MF, Bradley G, Lam DK. Topical 5-fluorouracil is a novel targeted therapy for the keratocystic odontogenic tumor. J Oral Maxillofac Surg. 2017;75(3):514-524. doi:10.1016/j.joms.2016.09.039.
10. Barua CG, Ali I, Tripathi A, Malakar A, Singha PK. The role of 5-fluorouracil in preventing recurrence after enucleation of odontogenic keratocyst: a case report. Cureus. 2023;15(9):e44777. doi:10.7759/cureus.44777.
11. Al-Moraissi EA, Kaur A, Gomez RS, Ellis E 3rd. Effectiveness of different treatments for odontogenic keratocyst: a network meta-analysis. Int J Oral Maxillofac Surg. 2023;52(1):32-43. doi:10.1016/j.ijom.2022.09.004.
12. Blanas N, Freund B, Schwartz M, Furst IM. Systematic review of the treatment and prognosis of the odontogenic keratocyst. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2000;90(5):553-558. doi:10.1067/moe.2000.110814.
13. Pogrel MA. Treatment of keratocysts: the case for decompression and marsupialization. J Oral Maxillofac Surg. 2005;63(11):1667-1673. doi:10.1016/j.joms.2005.08.008.

This work is licensed under a Creative Commons Attribution-NonCommercial-ShareAlike 4.0 International License.
Copyright (c) 2026 Rodrigo Javier Melara Estrada, Jennifer Alejandra Ramos Muñoz (Autor/a)

