scholarly journals Expect the unexpected – A rare case of Orthokeratinized Odontogenic Cyst and its surgical management

Author(s):  
M.M. Sheik Sameerudeen ◽  
◽  
R.N. Mugundan ◽  
S Shwetha ◽  
A. Fahmidha

Orthokeratinized Odontogenic Cyst (OOC) is a rare, developmental odontogenic cyst of the dental lamina. It was initially defined as the uncommon orthokeratinized variant of the Odontogenic Keratocyst (OKC), until the World Health Organization’s (WHO’s) classification in 2005 and 2017, where it was separated from the Keratocystic Odontogenic Tumor (KCOT) and has been included as a separate entity from the category of developmental odontogenic cysts respectively. It presents as a unilocular radiolucent lesion involving the posterior mandible and is frequently related to impacted teeth, often similar to other odontogenic cysts. Due to low local aggressiveness and less proliferative activity, it has to be differentiated from the other cysts in terms of surgical management. Here we report a rare case of OOC involving the maxilla along with an impacted canine and discuss the surgical management and why a secondary surgical intervention is unnecessary.

2012 ◽  
Vol 2 (2) ◽  
pp. 31-33 ◽  
Author(s):  
Niharika Swain ◽  
LS Poonja ◽  
Kamlesh Dekate

ABSTRACT Orthokeratinized odontogenic cyst (OOC) is a developmental cyst of odontogenic origin and was initially defined as the uncommon orthokeratinized variant of odontogenic keratocyst (OKC). However, recently World Health Organization has designated OOC as a distinct clinicopathologic entity as it has peculiar clinicopathologic aspects when compared to other developmental odontogenic cysts, especially OKCs. The orthokeratinized odontogenic cyst is histologically characterized by a thin, uniform, epithelial lining with orthokeratinization and a subjacent prominent granular cell layer. The purpose of the article is to present a case of OOC arising in the anterior mandible, an unusual site for the lesion and also highlights the importance of distinguishing it from the more commonly occurring keratocystic odontogenic tumor (KCOT). How to cite this article Swain N, Patel S, Poonja LS, Pathak J, Dekate K. Orthokeratinized Odontogenic Cyst. J Contemp Dent 2012;2(2):31-33.


2010 ◽  
Vol 134 (2) ◽  
pp. 271-275 ◽  
Author(s):  
Qing Dong ◽  
Shuang Pan ◽  
Li-Sha Sun ◽  
Tie-Jun Li

Abstract Context.—Orthokeratinized odontogenic cyst (OOC) is a relatively uncommon developmental cyst comprising about 10% of cases that had been previously coded as odontogenic keratocysts. Odontogenic keratocyst was designated as keratocystic odontogenic tumor (KCOT) in the new World Health Organization classification and OOC should be distinguished from KCOT for differences in histologic features and biologic behavior. Objective.—To analyze the clinicopathologic features of 61 cases of OOC in a Chinese population. Design.—Clinicopathologic analysis was performed on 61 cases of OOC. Immunohistochemical expression of Ki-67 and p63 was evaluated in 15 OOCs and 15 typical KCOTs. Results.—The 61 patients with OOC ranged from 13 to 75 years (average, 38.93 years). The lesions developed mainly in the third and fourth decades (57.38%) with a distinct predilection for males (72.13%). Six (9.84%) lesions were found in the maxilla and 55 (90.16%) in the mandible. The most common sites were in the mandibular molar and ramus region. Of the 54 cases with radiographic record, 47 (87.04%) were unilocular and 7 (12.96%) were multilocular radiolucencies. Twenty-seven of the 54 cysts were associated with an impacted tooth. Follow-up of 42 patients revealed no recurrence during an average period of 76.8 months after surgery. Compared with KCOTs, expression level of Ki-67 and p63 was significantly lower in OOCs, suggesting a lower proliferative activity. Conclusion.—Orthokeratinized odontogenic cyst is clinicopathologically distinct from KCOT and should constitute its own clinical entity.


2015 ◽  
Vol 6 (2) ◽  
pp. 40-43 ◽  
Author(s):  
Subhadeep Sarkar ◽  
Obaid Khursheed ◽  
Ruby Bansal ◽  
Waseem K Burza

2010 ◽  
Vol 41 (12) ◽  
pp. 1718-1725 ◽  
Author(s):  
Tadanobu Aragaki ◽  
Yasuyuki Michi ◽  
Ken-ichi Katsube ◽  
Narikazu Uzawa ◽  
Norihiko Okada ◽  
...  

2014 ◽  
Vol 6 (3) ◽  
pp. 23-27
Author(s):  
Heena Sonawane ◽  
Freny R Karjodkar ◽  
Kaustubh Sansare ◽  
Nimish Prakash

ABSTRACT Orthokeratinized odontogenic cyst (OOC) was first identified as the rare variant of keratocystic odontogenic tumor (KCOT) for its different histopathology and rare recurrence which was reclassified by WHO in 2005. The orthokeratinized odontogenic cyst is a distinct clinicopathologic entity and is histologically characterized by a thin, uniform, epithelial lining with orthokeratinization and a subjacent granular cell layer. The basal cells are usually cuboidal or flattened. OOC in maxilla is rare. This article presents a case of 56-years-old male patient with OOC in left maxilla. The clinical, radiographic and histological features of the cyst are discussed in this case report.


2013 ◽  
Vol 5 (3) ◽  
pp. 182-186
Author(s):  
Heena Sonawane ◽  
Freny R Karjodkar ◽  
Kaustubh Sansare ◽  
Nimish Prakash

ABSTRACT Orthokeratinized odontogenic cyst (OOC) was first identified as the rare variant of keratocystic odontogenic tumor (KCOT) for its different histopathology and rare recurrence which was reclassified by WHO in 2005. The orthokeratinized odontogenic cyst is a distinct clinicopathologic entity and is histologically characterized by a thin, uniform, epithelial lining with orthokeratinization and a subjacent granular cell layer. The basal cells are usually cuboidal or flattened. OOC in maxilla is rare. This article presents a case of 56-years-old male patient with OOC in left maxilla. The clinical, radiographic and histological features of the cyst are discussed in this case report. How to cite this article Sonawane H, Karjodkar FR, Sansare K, Prakash N. Orthokeratinized Odontogenic Cyst: A Rarity. Int J Otorhinolaryngol Clin 2013;5(3):182-186.


2020 ◽  
Vol 8 (11) ◽  
Author(s):  
Francielly Thomas Figueiredo ◽  
Alana Oswaldina Gavioli Meira Dos Santos ◽  
Julio Cesar Leite Da Silva ◽  
José Carlos Garcia De Mendonça ◽  
Gustavo Silva Pelissaro ◽  
...  

O queratocisto odontogênico é uma neoplasia benigna onde lesões únicas ou múltiplas são encontradas em ossos ou cavidades. O objetivo deste trabalho é relatar um caso clínico de uma lesão de queratocisto odontogênico, evidenciando corretos diagnóstico e terapêutica. O relato de caso trata-se de um paciente de 32 anos, sexo feminino, melanoderma, que deu entrada à faculdade de odontologia da Universidade Federal de Mato Grosso do Sul, no ano de 2017, apresentando lesão radiolúcida com halo esclerótico em região de ângulo de mandíbula do lado esquerdo, sem envolvimento de dentes e/ou raízes adjacentes. Foi realizada uma tomografia computadorizada, constando hipótese diagnóstica radiográfica da mencionada patologia. O plano de tratamento adotado preconizou uma intervenção conservadora de descompressão e acompanhamento pós-operatório. A paciente se encontra em pós-operatório de 19 meses cirúrgico sem queixas álgicas e/ou funcionais.Descritores: Cistos Ósseos; Descompressão; Terapêutica.ReferênciasShuster A, Shlomi B, Reiser V, Kaplan I. Solid keratocystic odontogenic tumor report of a non agressive case. J Oral Maxillofac Surg. 2012;70(4):865-70.Tsukamoto G, Sasaki A, Akiyama T, Ishikawa T, Kishimoto K, Nishiyama A, et al. A radiologic analysis of dentigerous cysts and odontogenic keratocysts associated with a mandibular third molar. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2001;91(6):743-47.Chan JKC, El-Naggar AK, Grandis JR, Takata T, Slootweg PJ. Who classification of head and neck tumours. World Health Organization 2017, 4th edition.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 Pathology. 2017; 11(1): 68-77.Gil JN, Rau Lh, Manfro R, Gasperini G, Dunker C, Chiarelli M. Ceratocisto odontogênico - caso clínico. Rev Port Estomatol Med Dent Cir Maxilofac. 2003;44(3):59-69.Regezi JÁ, Sciubba JJ. Patologia bucal: correlações clinicopatológicas. 3. ed. Rio de Janeiro: Guanabara Koogan; 2000.Larsen PE. Marsupialization for odontogenic keratocysts: Long-term follow-up analysis of the effects and changes in growth characteristics. 2002;94(5):543-53.Gambhir A, Rani G. Conservative management of keratocystic odontogenic tumour with enucleation, excision of the overlying mucosa and electrocauterization: a case report. West Indian Med J. 2014;63(7):775-78.Núñez-Urrutia S, Figueiredo R, Gay-Escoda C. Retrospective clinicopathological study of 418 odontogenic cysts. Med Oral Patol Oral Cir Bucal. 2010;15(5):e767-73.González-Alva P, Tanaka A, Oku Y, Yoshizawa D, Itoh S, Sakashita H, et al. Keratocystic odontogenic tumor: a retrospective study of 183 cases. J Oral Sci. 2008; 50(2):205-12.Jones AV, Craig GT, Franklin CD. Range and demographics of odontogenic cysts diagnosed in a UK population over a 30-year period. J Oral Pathol Med. 2006;35(8):500-7.Zhao YF, Wei JX, Wang SP. Treatment of odontogenic Keratocysts: a follow-up of 255 Chinese patients. Oral Surg Oral Med Oral Pathol Oral Radiol Endodo. 2002;94(2):151-56 .Meara JG, Shah S, Li KK, Cunningham MJ. The odontogenic keratocyst: a 20-year clinicophatologic review. Laryngoscope. 1998;108(2):280-83Kulkarni GH, Khaji SI, Metkari S, Kulkarni HS, Kulkarni R. Multiple keratocysts of the mandible in association with Gorlin-Goltz syndrome: A rare case report. Contemp Clin Dent. 2014;5(3):419-21.Speight PM, Takata T. New tumour entities in the 4th edition of the world health organization classification of head and neck tumours: odontogenic and maxillofacial bone tumours. Virchows Arch. 2018;472(3):331-39.Wright JM. The odontogenic keratocyst: orthokeratinized variant. Oral Surg. 1981;51(1):609-18.Hupp JR, Ellis III E, Tucker MR. Cirurgia oral e maxillofacial contemporânea. 5. ed. Rio de Janeiro: Elsevier; 2009.Dammer R, Niederdellmann H, Dammer P, Nuebler-Moritz M. Conservative or radical tretment of keratocysts: a retrospective review. Br J Oral Maxillofac Surg. 1997;35(1):46-8.Browne RM. The pathogeneses of odontogenic cysts: a review. J Oral Pathol. 1975;4(1):31-46.de Molon RS, Verzola MH, Pires LC, Mascarenhas VI, da Silva RB, Cirelli JA et al. Five years follow-up of a keratocyst odontogenic tumor treated by marsupialization and enucleation: A case report and literature review. Contemp Clin Dent. 2015;6(Suppl 1):S106-10.Neville BW, Damm DD, Allen CM, Bouquot JE. Patologia oral e maxilofacial. 3.ed. Rio de Janeiro: Elsevier; 2009.de Souza LB, Gordón-Núñez MAG, Nonaka CFW, de Medeiros MC, Torres TF, Emiliano GBG. Odontogenic cysts: Demographic profile in a Brazilian population over a 38-year period. Med Oral Patol Oral Cir Bucal. 2010;15(4):e583-90.EL-Gehani R, Orafi M, Elarbi M, Subhashraj K: Benign tumours of orofacial region at Benghazi, Libya: a study of 405 cases. J Craniomaxillofac Surg 2009;37(7):370-75.Moura BS, Cavalcante MA, Hespanhol W. Tumor odontogênico ceratocístico.  Keratocystic odontogenic tumor. Rev Col Bras Cir. 2016;43(6):466-71.Kaczmarzyk T, Mojsa I, Stypulkowska J. A systematic review of the recurrence rate for keratocystic odontogenic tumour in relation to treatment modalities. Int J Oral Maxillofac Surg. 2012;41(6):756-67.Lima GM, Nogueira RLM, Rabenhorst SHB. Considerações atuais sobre o comportamento biológico dos queratocistos odontogênicos. Rev Cir Traumatol Buco-Maxilo-fac. 2006;6(2):9-16.Madras J, Lapointe H. Keratocystic odontogenic tumour: reclassification of the odontogenic keratocyst from cyst to tumour. J Canad Dent Assoc. 2008; 74(2):165-165h.


2020 ◽  
Vol 35 (1) ◽  
pp. 76-77
Author(s):  
Jose Carnate

A 70-year-old man consulted for a mass of unspecified duration in the anterior area of the mandible. Radiologic information was not provided. An incision biopsy was performed by the dentistry service. The specimen received at the laboratory was labeled “cystic lining” and consisted of two light-gray to dark brown, irregularly shaped tissue fragments measuring 0.3 cm and 0.4 cm in diameters. Histological sections show biloculated cyst wall segments composed of fibrocollagenous tissue lined by an epithelial lining of varying thickness. (Figure 1) The latter consists of a thin layer of non-keratinizing epithelium with plaque-like thickenings that are composed of cells with a whorled pattern and variably clear cytoplasm. (Figures 2 & 3) Based on these microscopic features we signed the case out as a botryoid odontogenic cyst. Botryoid odontogenic cyst (BOC) is a developmental, non-inflammatory odontogenic cyst derived from residual odontogenic epithelium such as the dental lamina.1,2 It occurs between the roots of erupted teeth and is typically multilocular. It represents less than 1% of odontogenic cysts. Most cases occur in the sixth and seventh decades of life and a slight preponderance of males has been observed.1 Other studies report an equal distribution between sexes.2 It most often occurs in the mandible anterior to the molars, particularly the incisive/canine and premolar regions.1,2 Most BOCs are asymptomatic and discovered incidentally on radiographs although occasionally bone expansion is observed.1 Radiologically, BOCs often present with a multilocular radiological appearance.2 This multilocular feature has been likened to a “bunch of grapes” (botryoid: from the Greek botrys – bunch of grapes, and oeides – in the shape of).3 Microscopically, the cyst locules are lined by a one-to-two cell thick non-keratinizing epithelium with plaque-like thickenings of cells in a whorled arrangement and connective tissue septa.1,3 Cytoplasmic clearing may be observed because of glycogen.1 The differential diagnosis includes a lateral periodontal cyst (LPC) and a gingival cyst (GC). Although their histological features are largely identical, a LPC is unilocular while a GC occurs in the alveolar ridge of infants.1-3 A BOC is often considered a multilocular variant of lateral periodontal cyst.2,3 Making this distinction however is more than just of morphologic interest but is important as BOCs are reported to give rise to recurrences unlike LPCs and GCs.2,3 An ameloblastoma may be considered because of the multilocular appearance although the microscopic features are sufficiently distinct to rule out this entity. An adenomatoid odontogenic tumor (AOT) may be considered because of the nodular thickenings with a whorled pattern. However, AOT is a solid tumor characterized in addition by the presence of rosette- or duct-like spaces with dentinoid matrix. The recommended treatment is by enucleation.1 Successful conservative management with Carnoy’s solution has also been described.4 Peripheral ostectomy has also been recommended – a more aggressive approach being proposed as appropriate to extirpate any residual lesion.3 Recurrence has been stated to be between 15 to 30% which is largely ascribed to the multilocular characteristic of BOCs and consequent difficulty of complete removal, or of larger lesions.1-3 Thus, appropriate follow-up of cases or adjunctive therapy after enucleation might be warranted.2,3 To the author’s awareness, this is the first locally reported case of BOC after a search of local health research databases.


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