Abstract
The odontogenic keratocyst (OKC) is a benign lesion that arises from dental lamina remnants or basal cells of the overlying epithelium. In 2005, the World Health Organization categorized OKCs as tumors due to their aggressive behavior. However, in 2017 due to the lack of evidence, it was reclassified as an odontogenic cyst with aggressive behavior. This report describes a clinical case of a 20-year-old male who visited the dental clinic at the University of Costa Rica for third molar removal. As an incidental radiographic finding, an intraosseous lesion in the right side of the mandible was detected. The patient was asymptomatic, with an augmented mass in the right posterior sulcus and crepitation at palpation. Decompression, enucleation followed by peripheral ostectomy, and incisional biopsy were performed, and a personalized and handmade acrylic appliance was fitted into the surgical window until it closed. No recurrence has been documented after 6 years. The continuous debate regarding OKC’s classification by the WHO challenges the odontology community to constantly update its knowledge in the treatment modalities, taking into consideration the availability of resources and recurrence rates. The aim of this paper is to present the treatment and long-term follow-up of an OKC management and concomitantly review its main treatment options.
References
Li T.J. Critical reviews in oral biology & medicine: The odontogenic keratocyst: A cyst, or a cystic neoplasm? J Dent Res. 2011; 90 (2): 133-42.
Boffano P., Cavarra F., Agnone A.M., Brucoli M., Ruslin M., Forouzanfar T., et al. The epidemiology and management of odontogenic keratocysts (OKCs): A European multicenter study. J Cranio-Maxillofacial Surg. 2022; 50 (1): 1-6.
Pogrel M.A. The Keratocystic Odontogenic Tumor. Oral Maxillofac Surg Clin North Am. 2013 Feb; 25 (1): 21-30.
Carlson E.R., Oreadi D., McCoy J.M. Nevoid Basal Cell Carcinoma Syndrome and the Keratocystic Odontogenic Tumor. J Oral Maxillofac Surg [Internet]. 2015; 73 (12): S77-86. Available from: http://dx.doi.org/10.1016/j.joms.2015.04.037
Wright J.M., Vered M. Update from the 4th Edition of the World Health Organization Classification of Head and Neck Tumours: Odontogenic and Maxillofacial Bone Tumors. Head Neck Pathol. 2017; 11 (1): 68-77.
Soluk-Tekkesin M., Wright J.M. The World Health Organization Classification of Odontogenic Lesions: A Summary of the Changes of the 2022 (5th) Edition. Turk Patoloji Derg. 2022; 38 (2): 168-84.
Gagnier J.J., Kienle G., Altman D.G., Moher D., Sox H., Riley D.; the CARE Group. The CARE Guidelines: Consensus-based Clinical Case Reporting Guideline Development. BMJ Case Rep. 2013; doi: 10.1136/bcr-2013-201554 PMID: 24155002
Pogrel M.A., Jordan R.C.K. Marsupialization as a definitive treatment for the odontogenic keratocyst. J Oral Maxillofac Surg. 2004 Jun 1; 62 (6): 651-5.
MacDonald-Jankowski D.S. Orthokeratinized odontogenic cyst: A systematic review. Dentomaxillofacial Radiol. 2010 Dec; 39 (8): 455-67.
Da Silva M.J.A., De Sousa S.O.M., Corrêa L., Carvalhosa A.A., De Araújo V.C. Immunohistochemical study of the orthokeratinized odontogenic cyst: A comparison with the odontogenic keratocyst. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2002; 94 (6): 732-7.
Piattelli A. P53 protein expression in odontogenic cysts. J Endod. 2001; 27 (7): 459-61.
Boyne P.J., Hou D., Moretta C., Pritchard T. The multifocal nature of odontogenic keratocysts. J Calif Dent Assoc [Internet]. 2005 Dec [cited 2022 Oct 13]; 33 (12): 961-5. Available from: http://www.ncbi.nlm.nih.gov/pubmed/16454239
Shear M. The aggressive nature of the odontogenic keratocyst: Is it a benign cystic neoplasm? Part 2. Proliferation and genetic studies. Oral Oncol [Internet]. 2002 [cited 2022 Nov 5]; 38 (4): 323-31. Available from: https://pubmed.ncbi.nlm.nih.gov/12076694/
Stoelinga P.J.W. Long-term follow-up on keratocysts treated according to a defined protocol. Int J Oral Maxillofac Surg [Internet]. 2001 [cited 2022 Oct 13]; 30 (1): 14-25. Available from: https://pubmed.ncbi.nlm.nih.gov/11289615/
Chirapathomsakul D., Sastravaha P., Jansisyanont P. A review of odontogenic keratocysts and the behavior of recurrences. Oral Surg Oral Med Oral Pathol Oral Radiol Endod [Internet]. 2006 [cited 2022 Oct 13]; 101 (1): 5-9. Available from: https://pubmed.ncbi.nlm.nih.gov/16360602/
Shear M. Odontogenic keratocysts: clinical features. Oral Maxillofac Surg Clin North Am [Internet]. 2003 [cited 2022 Dec 4]; 15 (3): 335-45. Available from: https://pubmed.ncbi.nlm.nih.gov/18088687/
Shear M. The aggressive nature of the odontogenic keratocyst: Is it a benign cystic neoplasm? Part 1. Clinical and early experimental evidence of aggressive behaviour. Oral Oncol [Internet]. 2002 [cited 2022 Nov 5]; 38 (3): 219-26. Available from: https://pubmed.ncbi.nlm.nih.gov/11978543/
Kubota, Y., Ninomiya, T.,Oka, S., Takenoshita, Y., Shirasuna K. Interleukin- 1 a--dependent Regulation of Matrix Metalloproteinase-9 and Activation in the Epithelial Cells of Odontogenic Jaw Cysts. J Dent RES [Internet]. 2000; 79 (6): 1423-30. Available from: https://pubmed.ncbi.nlm.nih.gov/10890723/
Bhargava D, Deshpande A, Pogrel MA. Keratocystic odontogenic tumour (KCOT)-a cyst to a tumour. Oral Maxillofac Surg. 2012 Jun; 16 (2): 163-70.
Kinard B., Hansen G., Newman M., Dennis P., Haeffs T., Perez S., et al. How well do we manage the odontogenic keratocyst? A multicenter study. Oral Surg Oral Med Oral Pathol Oral Radiol. 2019 Apr 1; 127 (4): 282-8.
Myoung H., Hong S.P., Hong S.D., Lee J. Il, Lim C.Y., Choung P.H, et al. Odontogenic keratocyst: Review of 256 cases for recurrence and clinicopathologic parameters. Oral Surg Oral Med Oral Pathol Oral Radiol Endod [Internet]. 2001 [cited 2022 Nov 23]; 91 (3): 328-33. Available from: https://pubmed.ncbi.nlm.nih.gov/11250631/
Boiidar Pavelić, Marina Katunarić, Sanja Segović, Maja Cimas Karadole, Davor Katanec, Aida Saban IP. The incidence of satellite cysts in keratocystic odontogenic tumors.: EBSCOhost [Internet]. JOURNAL OF THE CROATION ANTHROPOLOGICAL SOCIETY. 2014 [cited 2022 Nov 10]. p. Vol 38. 269-273. Available from: https://web-s-ebscohost-com.ezproxy.sibdi.ucr.ac.cr/ehost/pdfviewer/pdfviewer?vid=0&sid=e2bcca97-9577-4625-b34f-0a1a2ed18ce2%40redis
Raj A., Bahadur R., Bansal S., Kumar P. Intricate correlation and biological behavior of keratocyst in nevoid basal cell carcinoma syndrome: A comprehensive review of literature. Clin Cancer Investig J [Internet]. 2013 [cited 2022 Nov 23]; 2 (3): 185. Available from: https://ccij-online.org/article/intricate-correlation-and-biological-behavior-of-keratocyst-in-nevoid-basal-cell-carcinoma-syndrome:-a-comprehensive-review-of-literature-113
Zhao Y., Liu B., Cheng G., Wang S.P., Wang Y.N. Recurrent keratocystic odontogenic tumours: Report of 19 cases. Dentomaxillofacial Radiol. 2012 Feb 1; 41 (2): 96-102.
Fidele N.B., Yueyu Z., Zhao Y., Tianfu W., Liu J., Sun Y., et al. Recurrence of odontogenic keratocysts and possible prognostic factors: Review of 455 patients. Med Oral Patol Oral y Cir Bucal. 2019 Jul 1; 24 (4): e491-501.
Morgan T.A., Burton C.C., Qian F. A retrospective review of treatment of the odontogenic keratocyst. J Oral Maxillofac Surg. 2005; 63 (5): 635-9.
Bakaeen G., Rajab L.D., Sawair F.A., Hamdan M.A.M., Dallal N.D. Nevoid basal cell carcinoma syndrome: a review of the literature and a report of a case. Int J Paediatr Dent [Internet]. 2004 Jul [cited 2023 Jan 20]; 14 (4): 279-87. Available from: https://pubmed.ncbi.nlm.nih.gov/15242385/
Balasundram S., Kovilpillai F.J., Hopper C. Nevoid basal cell carcinoma syndrome presenting with neck pits and café au lait patches. J Clin Pediatr Dent [Internet]. 2010 Sep 1 [cited 2022 Oct 13]; 35 (1): 95-100. Available from: http://www.ncbi.nlm.nih.gov/pubmed/21189772
Maurette P.E., Jorge J., De Moraes M. Conservative treatment protocol of odontogenic keratocyst: a preliminary study. J Oral Maxillofac Surg [Internet]. 2006 [cited 2023 Jan 17]; 64 (3): 379-83. Available from: https://pubmed.ncbi.nlm.nih.gov/16487797/
Kawai T., Murakami S., Hiranuma H., Sakuda M. Healing after removal of benign cysts and tumors of the jaws. A radiologic appraisal. Oral Surgery, Oral Med Oral Pathol Oral Radiol. 1995; 79 (4): 517-25.
Bodner L., Bar-Ziv J. Characteristics of bone formation following marsupialization of jaw cysts. Dentomaxillofacial Radiol. 1998; 27 (3): 166-71.
MacDonald-Jankowski D.S. Keratocystic odontogenic tumour: Systematic review. Dentomaxillofacial Radiol. 2011 Jan; 40 (1): 1-23.
Kolokythas A., Fernandes R.P., Pazoki A., Ord RA. Odontogenic Keratocyst: To Decompress or Not to Decompress? A Comparative Study of Decompression and Enucleation Versus Resection/Peripheral Ostectomy. J Oral Maxillofac Surg. 2007 Apr; 65 (4): 640-4.
de Castro M.S., Caixeta C.A., de Carli M.L., Ribeiro Júnior N.V., Miyazawa M., Pereira A.A.C., et al. Conservative surgical treatments for nonsyndromic odontogenic keratocysts: a systematic review and meta-analysis. Clin Oral Investig. 2018 Jun 1; 22 (5): 2089-101.
Madras, Jonatha; Lapointe H. Keratocystic odontogenic tumour: reclassification of the odontogenic keratocyst from cyst to tumour - PubMed. J Can Dent Assoc [Internet]. 2008 [cited 2023 Jan 17]; 74 (2): 165-165h. Available from: https://pubmed.ncbi.nlm.nih.gov/18353202/
Giuliani M., Grossi G.B., Lajolo C., Bisceglia M., Herb K.E. Conservative management of a large odontogenic keratocyst: report of a case and review of the literature. J Oral Maxillofac Surg [Internet]. 2006 Feb [cited 2023 Jan 21]; 64 (2): 308-16. Available from: https://pubmed.ncbi.nlm.nih.gov/16413905/
Zhao Y.F., Wei J.X., Wang S.P. Treatment of odontogenic keratocysts: A follow-up of 255 Chinese patients. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2002; 94 (2): 151-6.
Dashow J.E., McHugh J.B., Braun T.M., Edwards S.P., Helman J.I., Ward B.B. Significantly decreased recurrence rates in keratocystic odontogenic tumor with simple enucleation and curettage using carnoy’s versus modified carnoy’s solution. J Oral Maxillofac Surg [Internet]. 2015 Nov 1 [cited 2022 Oct 13]; 73 (11): 2132-5. Available from: http://dx.doi.org/10.1016/j.joms.2015.05.005
Blanas N., Freund B., Frcd C., Schwartz M. Systematic review of the treatment and prognosis of the odontogenic keratocyst. Oral Surg Oral Med Oral Pathol Oral Radiol Endod [Internet]. 2000; 90 (5): 553-8. Available from: https://pubmed.ncbi.nlm.nih.gov/11077375/
Boffano P., Ruga E. Keratocystic odontogenic tumor (odontogenic keratocyst): preliminary retrospective review of epidemiologic, clinical, and radiologic features of 261 lesions from University of Turin. J Oral Maxillofac Surg [Internet]. 2010; 68 (12): 2994-9. Available from: https://pubmed.ncbi.nlm.nih.gov/20970907/
Pogrel M.A. Treatment of keratocysts: The case for decompression and marsupialization. J Oral Maxillofac Surg. 2005 Nov; 63 (11): 1667-73.
Johnson N.R., Batstone M.D., Savage N.W. Management and recurrence of keratocystic odontogenic tumor: A systematic review. Oral Surg Oral Med Oral Pathol Oral Radiol [Internet]. 2013; 116 (4): e271-6. Available from: http://dx.doi.org/10.1016/j.oooo.2011.12.028
Ninomiya T, Kubota Y, Koji T, Shirasuna K. Marsupialization inhibits interleukin-1alpha expression and epithelial cell proliferation in odontogenic keratocysts. J Oral Pathol Med [Internet]. 2002 Oct [cited 2022 Nov 5]; 31 (9): 526-33. Available from: https://pubmed.ncbi.nlm.nih.gov/12269991/
Tolstunov L., Treasure T. Surgical Treatment Algorithm for Odontogenic Keratocyst: Combined Treatment of Odontogenic Keratocyst and Mandibular Defect With Marsupialization, Enucleation, Iliac Crest Bone Graft, and Dental Implants. J Oral Maxillofac Surg. 2008 May; 66 (5): 1025-36.
Slusarenko da Silva Y., Stoelinga P.J.W., Naclério-Homem M. da G. Recurrence of nonsyndromic odontogenic keratocyst after marsupialization and delayed enucleation vs. enucleation alone: a systematic review and meta-analysis. Oral Maxillofac Surg. 2019 Mar 6; 23 (1): 1-11 .
Cunha J.F., Gomes C.C., De Mesquita R.A., Andrade Goulart E.M., De Castro W.H., Gomez R.S. Clinicopathologic features associated with recurrence of the odontogenic keratocyst: A cohort retrospective analysis. Oral Surg Oral Med Oral Pathol Oral Radiol. 2016 Jun 1; 121 (6): 629-35.
Donnelly L.A., Simmons T.H., Blitstein B.J., Pham M.H., Saha P.T., Phillips C., et al. Modified Carnoy’s Compared to Carnoy’s Solution Is Equally Effective in Preventing Recurrence of Odontogenic Keratocysts. J Oral Maxillofac Surg. 2021 Sep 1; 79 (9): 1874-81.
Motaleb L., Zakai D., Stocker J. Fourteen-year study of the management of the odontogenic keratocyst. Are adjunctive therapies all they are cut out to be? Br J Oral Maxillofac Surg. 2022 Feb 1; 60 (2): 105-12.
Schmidt B.L., Pogrel M.A. The use of enucleation and liquid nitrogen cryotherapy in the management of odontogenic keratocysts. J Oral Maxillofac Surg [Internet]. 2001 [cited 2022 Oct 13]; 59 (7): 720-5. Available from: https://pubmed.ncbi.nlm.nih.gov/11429726/
Ribeiro-Júnior O., Borba A.M., Alves C.A.F., de GOuveia M.M., Deboni M.C.Z., da Graça Naclério-Homem M. Reclassification and treatment of odontogenic keratocysts: A cohort study. Braz Oral Res. 2017; 31 (1): 1-10.
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