Surgical Treatment for Multiple Odontogenic Keratocyst : A Case Report

Authors

  • Alisara Ngerncham Uthaithani Hospital

Keywords:

Odontogenic Keratocyst, Nevoid basal cell carcinoma syndrome

Abstract

A 21-year-old female was referred to the Dental Department, Uthaithani Hospital, with multiple cystic lesions in the maxilla and mandible observed on a panoramic radiograph. The patient visited an orthodontist complaining of the failure of permanent teeth to erupt. Extraoral examination revealed multiple palmar pits. Intraoral examination showed a partly edentulous state; teeth 18, 26, 28, 37, 38, 46, and 48 were missing. A panoramic radiograph revealed multiple large radiolucencies with well-defined margins in the posterior maxilla and mandible. A skull radiograph revealed calcification of the falx cerebri. Marsupialization and biopsy of cystic lesions in the right and left mandible were performed under local anesthesia. The histopathologic report confirmed the diagnosis of odontogenic keratocyst (OKC). Based on all clinical, radiographic, and histopathologic findings, this case suggested the possibility of nevoid basal cell carcinoma syndrome (NBCCS). After 10 months of marsupialization, the patient was admitted to the hospital, and enucleation of all cystic lesions was performed under general anesthesia. There were no postoperative complications and no signs of recurrence after a 2-year follow-up.

Keywords: Odontogenic Keratocyst, Nevoid basal cell carcinoma syndrome

References

Philipsen HP. Om keratocyster (kolesteratomer) i kaeberne. Tandlaegebladet. 1956;60:963–71.

Barnes L, Eveson JW, Sidransky D, Reichart P, editors. Pathology and genetics of head and neck tumours.9th ed. Lyon: IARC press; 2005.

Madras J, Lapointe H. Keratocystic odontogenic tumour: reclassification of the odontogenic keratocyst from cyst to tumour. J Can Dent Assoc. 2008;74(2):165.

Wright JM, 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.

Simiyu BN, Butt F, Dimba EA, Wagaiyu EG, Awange DO, Guthua SW, et al. Keratocystic odontogenic tumours of the jaws and associated pathologies: a 10 year clinicopathologic audit in a referral teaching hospital in Kenya. J Craniomaxillofac Surg. 2013;41(3):230-4.

Neville BW, Damm DD, Allen CM, Bouquo JE. Oral and maxillofacial pathology, 2nd ed. Philadelphia : WB Saunders; 2002.

Hasheminia D, Naemi V, and Naghdi N. Conservative treatment protocol of keratocystic odontogenic tumor : report of a case with Nevoid basal cell carcinoma syndrome and literature review. J Maxillofac Oral Surg. [internet] 2015 Mar [cited 2021 Sep 14];14(1):475-81. Available from:http://www.pubmed.ncbi.nlm.nih.gov/25848161/

Ortega Garcia de Amezaga A, Garcia Arregui O, Zepeda Nuno S, Acha Sagredo A, Aguirre Urizar JM. Gorlin-Goltz syndrome : clinicopathologic aspects. Med Oral Patol Oral Cir Bucal. 2008;13:338-43

Kimonis VE, Goldstein AM, Pastakia B, Yang ML, Kase R, Digiovanna JJ, et al. Clinical manifestrations in 105 persons with nevoid basal cell carcinoma syndrome. Am J Med Genet. 1997;69:299-308.

The American Academy of otolaryngology head and neck surgery foundation. Resident manual of trauma to the face, head and neck. [internet] First edition 2012.[cite 2021 Sep 25]. Available from: https://www.entnet.org.

Lindeboom JA, Kroon FH, de Vires J, van den Akker HP. Multiple recurrent and de novo odontogenic keratocysts associated with oral-facialdigital syndrome. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2003;95(4):458–62.

Dominguez FV, Keszler A. Comparative study of keratocysts, associated and non-associated with nevoid basal cell carcinoma syndrome. J Oral Pathol. 1988;17(1):39–42.

Kavitha EG, Jayanthi P, Senthilmoorthy, Rathy R, harish RK, Ameena M. Bilateral odontogenic keratocyst in a nonsyndromic patient : a case report and review of literature. Oral Maxillofac Pahol J. 2019;10:30-4.

Gorlin syndrome alliance. About Gorlin syndrome [internet]. 2021. [cited 2021 sep 16]. Available from: https://gorlinsyndrome.org.

Golgire SM, Varekar AA, Patil A, Shetti SS, and Magdum D. Non-syndromic multiple odontogenic keratocysts : a case report and comparison with syndromic multiple odontogenic keratocysts. J clin Case Rep. 2016;6:6.

Farndon PA, Del Mastro RG, Evans DG, Kilpatrick MW. Location of gene for Gorlin syndrome. Lancet. 1992;339:581-2.

Kopera D, Cerroni L, Fink-Puches R, Kerl H. Different treatment modalities for the management of a patient with the nevoid basal cell carcinoma syndrome. J Am Acad Dermadol. 1996;34:937-9.

Blanas N, Freund B, Schwartz M, Furst M. Systematic review of the treatment and prognosis of the odontogenic keratocyst. Oral surg Oral Med Oral Pathol Oral Radiol Endod. 2000;90:553-8.

Forssell K, Sainio P. Clinicopathological study of keratinized cysts of the jaws. Proc Finn Dent Soc. 1979;75:36-45.

Published

2025-08-29

How to Cite

เงินแจ่ม อลิสรา. 2025. “Surgical Treatment for Multiple Odontogenic Keratocyst : A Case Report”. Region 3 Medical and Public Health Journal - วารสารวิชาการแพทย์และสาธารณสุข เขตสุขภาพที่ 3 22 (3). Nakhonsawan Thailand:280-87. https://thaidj.org/index.php/smj/article/view/16594.

Issue

Section

รายงานผู้ป่วย (Case Report)