Giant Keratocystic Odontogenic Tumor: Three Cases and Literature Review

Overview

Journal Iran J Otorhinolaryngol

Publisher Mashhad University of Medical Sciences

Specialty Otorhinolaryngology

Date 2013 Dec 5

PMID 24303448

Citations 5

Authors

Alexandre Caixeta Guimaraes

Mariana Dutra de Cassia Ferreira Santos

Guilherme Machado de Carvalho

Carlos Takahiro Chone

Leopoldo Nizam Pfeilsticker

Affiliations

Soon will be listed here.

Abstract

Introduction: A keratocystic odontogenic tumor is a benign intra-bone mass originating from dental lamina or its residue. It represents 2-11% of jaw cysts, and has a slow but aggressive growth. The evaluation of molecular characteristics, immunohistochemistry, and genetic expression currently have no established classification regarding the evolution and pathophysiologic pattern of these lesions.

Case Report: This is a clinical retrospective study with a full analysis of patient history regarding physical evaluation, radiologic images, pathology results, and surgical resection. We performed a major literature review concerning current concepts relating to its biological characterization. Three cases of keratocystic odontogenic tumor were identified. Two of the cases were large, with aggressive behavior and significant bone destruction and recurrence, which had been overlooked for more than a decade. The third case had an early diagnosis, and the treatment led to full recovery and complete healing.

Conclusion: The keratocystic odontogenic tumor is a benign lesion with slow growth, which lends itself to a more conservative treatment, even in cases of large lesions. A better understanding of these tumors, both at the biological and molecular level, could lead to guidelines for treatment and prognosis of such patients.

Citing Articles

Treatment For The Large Aggressive Benign Lesions Of The Jaws.

Cakarer S, Isler S, Keskin B, Uzun A, Berberoglu H, Keskin C J Maxillofac Oral Surg. 2018; 17(3):372-378.

PMID: 30034157 PMC: 6028335. DOI: 10.1007/s12663-017-1060-5.

Big Keratocystic Odontogenic Tumor of the Mandible: A Case Report.

Kebede B, Dejene D, Teka A, Girma B, Aguirre E, Guerra N Ethiop J Health Sci. 2017; 26(5):491-496.

PMID: 28446856 PMC: 5389065. DOI: 10.4314/ejhs.v26i5.12.

Giant keratocystic odontogenic tumor of the mandible - a case report.

Kornafel O, Jazwiec P, Pakulski K Pol J Radiol. 2015; 79:498-501.

PMID: 25566331 PMC: 4280059. DOI: 10.12659/PJR.892048.

Rare and massive odontogenic parakeratotic cyst treated by endoscopic sinus surgery: a case report.

Marcotullio D, Iannella G, Zelli M, Marinelli C, Magliulo G J Med Case Rep. 2014; 8:293.

PMID: 25193270 PMC: 4164324. DOI: 10.1186/1752-1947-8-293.

Bimaxillary keratocystic odontogenic tumour: a case of diagnostic and therapeutic difficulty.

Okoje-Adesomoju V, Adisa A, Gbolahan O, Olajide M Case Rep Med. 2014; 2014:194810.

PMID: 24790606 PMC: 3984809. DOI: 10.1155/2014/194810.

References

Sharif F, Oliver R, Sweet C, Sharif M . Interventions for the treatment of keratocystic odontogenic tumours (KCOT, odontogenic keratocysts (OKC)). Cochrane Database Syst Rev. 2010; (9):CD008464. DOI: 10.1002/14651858.CD008464.pub2. View

Kuroyanagi N, Sakuma H, Miyabe S, Machida J, Kaetsu A, Yokoi M . Prognostic factors for keratocystic odontogenic tumor (odontogenic keratocyst): analysis of clinico-pathologic and immunohistochemical findings in cysts treated by enucleation. J Oral Pathol Med. 2009; 38(4):386-92. DOI: 10.1111/j.1600-0714.2008.00729.x. View

Morgan T, Burton C, Qian F . A retrospective review of treatment of the odontogenic keratocyst. J Oral Maxillofac Surg. 2005; 63(5):635-9. DOI: 10.1016/j.joms.2004.07.026. View

Falaki F, Delavarian Z, Salehinejad J, Saghafi S . Squamous cell carcinoma arising from an odontogenic keratocyst: a case report. Med Oral Patol Oral Cir Bucal. 2009; 14(4):E171-4. View

August M, Faquin W, Troulis M, Kaban L . Dedifferentiation of odontogenic keratocyst epithelium after cyst decompression. J Oral Maxillofac Surg. 2003; 61(6):678-83; discussion 683-4. DOI: 10.1053/joms.2003.50137. View

Sembronio S, Albiero A, Zerman N, Costa F, Politi M . Endoscopically assisted enucleation and curettage of large mandibular odontogenic keratocyst. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2008; 107(2):193-6. DOI: 10.1016/j.tripleo.2008.07.020. View

Zhang L, Sun Z, Zhao Y, Bian Z, Fan M, Chen Z . Inhibition of SHH signaling pathway: molecular treatment strategy of odontogenic keratocyst. Med Hypotheses. 2006; 67(5):1242-4. DOI: 10.1016/j.mehy.2006.04.062. View

Aragaki T, Michi Y, Katsube K, Uzawa N, Okada N, Akashi T . Comprehensive keratin profiling reveals different histopathogenesis of keratocystic odontogenic tumor and orthokeratinized odontogenic cyst. Hum Pathol. 2010; 41(12):1718-25. DOI: 10.1016/j.humpath.2010.05.007. View

Ihan Hren N, Miljavec M . Spontaneous bone healing of the large bone defects in the mandible. Int J Oral Maxillofac Surg. 2008; 37(12):1111-6. DOI: 10.1016/j.ijom.2008.07.008. View

10.

Zecha J, Amaral Mendes R, Lindeboom V, van der Waal I . Recurrence rate of keratocystic odontogenic tumor after conservative surgical treatment without adjunctive therapies - A 35-year single institution experience. Oral Oncol. 2010; 46(10):740-2. DOI: 10.1016/j.oraloncology.2010.07.004. View

11.

Pitak-Arnnop P, Chaine A, Oprean N, Dhanuthai K, Bertrand J, Bertolus C . Management of odontogenic keratocysts of the jaws: a ten-year experience with 120 consecutive lesions. J Craniomaxillofac Surg. 2009; 38(5):358-64. DOI: 10.1016/j.jcms.2009.10.006. View

12.

Li T . The odontogenic keratocyst: a cyst, or a cystic neoplasm?. J Dent Res. 2011; 90(2):133-42. DOI: 10.1177/0022034510379016. View

13.

Ruhin-Poncet B, Picard A, Martin-Duverneuil N, Albertini A, Goudot P . [Keratocysts (or keratocystic epithelial odontogenic tumors)]. Rev Stomatol Chir Maxillofac. 2011; 112(2):87-92. DOI: 10.1016/j.stomax.2011.01.010. View

14.

Boyne P, Hou D, Moretta C, Pritchard T . The multifocal nature of odontogenic keratocysts. J Calif Dent Assoc. 2006; 33(12):961-5. View

15.

Zhao Y, Liu B, Wang S, Wang Y . Computed densitometry of panoramic radiographs in evaluation of bone healing after enucleation of mandibular odontogenic keratocysts. Chin J Dent Res. 2011; 13(2):123-6. View

16.

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

17.

Amaral Mendes R, Carvalho J, van der Waal I . Biological pathways involved in the aggressive behavior of the keratocystic odontogenic tumor and possible implications for molecular oriented treatment - an overview. Oral Oncol. 2009; 46(1):19-24. DOI: 10.1016/j.oraloncology.2009.10.009. View

18.

Amaral Mendes R, Carvalho J, van der Waal I . Characterization and management of the keratocystic odontogenic tumor in relation to its histopathological and biological features. Oral Oncol. 2010; 46(4):219-25. DOI: 10.1016/j.oraloncology.2010.01.012. View