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Review Paper Clinical Pathology| Volume 48, ISSUE 11, P1405-1410, November 2019

Clear cell odontogenic carcinoma: a rare jaw tumor. A summary of 107 reported cases

      Abstract

      The purpose of this study was to summarize the currently published cases of clear cell odontogenic carcinoma (CCOC). The PubMed and Springer databases were used to collect available reports, searching for ‘clear cell odontogenic carcinoma’, ‘CCOC’, or ‘clear cell ameloblastoma’. The search resulted in 75 reports detailing 107 cases between 1985 and 2018. Clinically the tumor manifests as a swelling in the posterior mandible (n = 46), anterior mandible (n = 33), and maxilla (n = 28). Radiological analysis of 85 cases typically showed a poorly defined expansive radiolucency (n = 83). Of the 70 patients with symptoms reported, 44 specified a swelling, 11 tooth mobility, seven gingival/periodontal issues, five numbness, and three decreased jaw opening. One patient presented with a neck mass. The duration of symptoms prior to seeking care was specified for 52 patients: 2 months to 1 year for 34 patients, 1–2 years for seven, 2–4 years for two, 4–7 years for six, and 7–12 years for three. The incidence of recurrence appeared to be 38 of the 88 cases where recurrence was reported. CCOC can be distinguished from other oral cancers by its distinctive histology and immunohistochemical characteristics and less aggressive behavior. Currently, treatment should be early and aggressive resection with clear surgical margins and long-term follow-up. The overall goal is to collect a cohort of patients.

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      References

        • Hansen L.S.
        • Eversole R.L.
        • Green T.L.
        • Powell N.B.
        Clear cell odontogenic tumor—a new histologic variant with aggressive potential.
        Head Neck Surg. 1985; 8: 115-123
        • Kwon I.J.
        • Kim S.M.
        • Amponsah E.K.
        • Myoung H.
        • Lee J.H.
        • Lee S.K.
        Mandibular clear cell odontogenic carcinoma.
        World J Surg Oncol. 2015; 13: 284
        • Loyola A.M.
        • Cardoso S.V.
        • Rogerio de Faria P.
        • Servato J.P.S.
        • de Paulo L.F.B.
        • Eisenberg A.L.A.
        • Dias F.L.
        • Gomes C.C.
        • Gomez R.S.
        Clear cell odontogenic carcinoma: report of 7 new cases and systematic review of the current knowledge.
        Oral Surg Oral Med Oral Pathol Oral Radiol. 2015; 120: 483-496
        • Panda S.
        • Sahoo S.R.
        • Srivastav Padhiary S.
        • Dhull K.S.
        • Aggarwal S.
        Pathogenesis and nomenclature of odontogenic carcinomas: revisited.
        J Oncol. 2014; 2014197425
        • Hadj Saïd M.
        • Ordioni U.
        • Benat G.
        • Gomez-Brouchet A.
        • Chossegros C.
        • Catherine J.H.
        Clear cell odontogenic carcinoma. A review.
        J Stomatol Oral Maxillofac Surg. 2017; 118: 363-370
        • Bilodeau E.A.
        • Weinreb I.
        • Antonescu C.R.
        • Zhang L.
        • Dacic S.
        • Muller S.
        • Barker B.
        • Seethala R.R.
        Clear cell odontogenic carcinomas show EWSR1 rearrangements: a novel finding and a biological link to salivary clear cell carcinomas.
        Am J Surg Pathol. 2013; 37: 1001-1005
      1. Troulis MJ, Rivera MN, Faquin WC, Iafrate AJ. Unpublished data.

        • August M.
        • Faquin Troulis M.
        • Kaban L.
        Clear cell odontogenic carcinoma: evaluation of reported cases.
        J Oral Maxillofac Surg. 2003; 61: 580-586
        • Waldron C.A.
        • Small I.A.
        • Silverman H.
        Clear cell ameloblastoma—an odontogenic carcinoma.
        J Oral Maxillofac Surg. 1985; 43: 707-717
        • Muller H.
        • Slootweg P.
        Clear cell differentiation in an ameloblastoma.
        J Maxillofac Surg. 1986; 14: 158-160
        • Bang G.
        • Koppang H.S.
        • Gilhuus-Moe O.
        • Aksdal E.
        • Persson P.G.
        • Lundgren J.
        Clear cell odontogenic carcinoma: report of three cases with pulmonary and lymph node metastases.
        J Oral Pathol Med. 1989; 18: 113-118
        • Ng K.H.
        • Siar C.H.
        Peripheral ameloblastoma with clear cell differentiation.
        Oral Surg Oral Med Oral Pathol. 1990; 70: 210-213
        • Guilbert F.
        • Auriol M.
        • Chomette G.
        An unusual form of primary epithelioma of the mandible: odontogenic clear cell carcinoma. Clinical and morphologic study.
        Rev Stomatol Chir Maxillofac. 1991; 92: 277-280
        • Fan J.
        • Kubota E.
        • Imamura H.
        • Shimokama T.
        • Tokunaga O.
        • Katsuki T.
        • Watanabe T.
        Clear cell odontogenic carcinoma. A case report with massive invasion of neighboring organs and lymph node metastasis.
        Oral Surg Oral Med Oral Pathol. 1992; 74: 768-775
        • Odukoya O.
        • Arole O.
        Clear-cell ameloblastoma of the mandible (a case report).
        Int J Oral Maxillofac Surg. 1992; 21: 358-359
        • Milles M.
        • Doyle J.
        • Mesa M.
        • Raz S.
        Clear cell odontogenic carcinoma with lymph node metastasis.
        Oral Surg Oral Med Oral Pathol. 1993; 76: 82-89
        • Piattelli A.
        • Sesenna E.
        • Trisi P.
        Clear cell odontogenic carcinoma. Report of a case with lymph node and pulmonary metastases.
        Eur J Cancer B Oral Oncol. 1994; 30B: 278-280
        • Sadeghi E.M.
        • Levin S.
        Clear cell odontogenic carcinoma of the mandible: report of a case.
        J Oral Maxillofac Surg. 1995; 53: 613-616
        • Eversole L.R.
        • Duffey D.C.
        • Powell N.B.
        Clear cell odontogenic carcinoma. A clinicopathologic analysis.
        Arch Otolaryngol Head Neck Surg. 1995; 121: 685-689
        • Mari A.
        • Escutia E.
        • Carrera M.
        • Pericot J.
        Clear cell ameloblastoma or odontogenic carcinoma. A case report.
        J Craniomaxillofac Surg. 1995; 23: 387-390
        • Ferreira de Aguiar M.C.
        • Gomez R.S.
        • Silva E.C.
        • Cavalcanti de Araujo V.
        Clear-cell ameloblastoma (clear-cell odontogenic carcinoma): report of a case.
        Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1996; 81: 79-83
        • Muramatsu T.
        • Hashimoto S.
        • Inoue T.
        • Shimono M.
        • Noma H.
        • Shigematsu T.
        Clear cell odontogenic carcinoma in the mandible: histochemical and immunohistochemical observations with a review of the literature.
        J Oral Pathol Med. 1996; 25: 516-521
        • Yamamoto H.
        • Inui M.
        • Mori A.
        • Tagawa T.
        Clear cell odontogenic carcinoma: a case report and literature review of odontogenic tumors with clear cells.
        Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1998; 86: 86-89
        • Miyauchi M.
        • Ogawa I.
        • Takata T.
        • Ito H.
        • Nikai H.
        • Ijuhin N.
        • Tanimoto K.
        • Itoh Y.
        Clear cell odontogenic tumour: a case with induction of dentin-like structures?.
        J Oral Pathol Med. 1998; 27: 220-224
        • Kumamoto H.
        • Kawamura H.
        • Ooya K.
        Clear cell odontogenic tumor in the mandible: report of a case with an immunohistochemical study of epithelial cell markers.
        Pathol Int. 1998; 48: 618-622
        • Kumamoto H.
        • Yamazaki S.
        • Sato A.
        • Yamaguchi T.
        • Tezuka F.
        • Ooya K.
        Clear cell odontogenic tumor in the mandible: report of a case with duct-like appearances and dentinoid induction.
        J Oral Pathol Med. 2000; 29: 43-47
        • Nair M.K.
        • Burkes J.
        • Chai-U-Dom O.
        Radiographic manifestation of clear cell odontogenic tumor.
        Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2000; 89: 250-254
        • Benton D.C.
        • Eisenberg E.
        Clear cell odontogenic carcinoma: report of a case.
        J Oral Maxillofac Surg. 2001; 59: 83-88
        • Brinck U.
        • Gunawan B.
        • Schulten H.J.
        • Pinzon W.
        • Fischer U.
        • Füzesi L.
        Clear-cell odontogenic carcinoma with pulmonary metastases resembling pulmonary meningothelial-like nodules.
        Virchows Arch. 2001; 438: 412-417
        • Maiorano E.
        • Altini M.
        • Viale G.
        • Piattelli A.
        • Favia G.
        Clear cell odontogenic carcinoma. Report of two cases and review of the literature.
        Am J Clin Pathol. 2001; 116: 107-114
        • Li T.J.
        • Yu SF Gao Y.
        • Wang E.B.
        Clear cell odontogenic carcinoma: a clinicopathologic and immunocytochemical study of 5 cases.
        Arch Pathol Lab Med. 2001; 125: 1566-1571
        • Iezzi G.
        • Rubini C.
        • Fioroni M.
        • Piattelli A.
        Clear cell odontogenic carcinoma.
        Oral Oncol. 2002; 38: 209-213
        • Adamo A.K.
        • Boguslaw B.
        • Coomaraswarmy M.A.
        • Simos C.
        Clear cell odontogenic carcinoma of the mandible: case report.
        J Oral Maxillofac Surg. 2002; 60: 121-126
        • Ariyoshi Y.
        • Shimahara M.
        • Miyauchi M.
        • Nikai H.
        Clear cell odontogenic carcinoma with ghost cells and inductive dentin formation—report of a case in the mandible.
        J Oral Pathol Med. 2002; 31: 181-183
        • Mosqueda-Taylor A.
        • Meneses-Garcia A.
        • Ruiz-Godoy Rivera L.M.
        • De Lourdes Suarez-Roa M.
        Clear cell odontogenic carcinoma of the mandible.
        J Oral Pathol Med. 2002; 31: 439-441
        • Brandwein M.
        • Said-Al-Naeif N.
        • Gordon R.
        • Urken M.
        Clear cell odontogenic carcinoma: report of a case and analysis of the literature.
        Arch Otolaryngol Head Neck Surg. 2002; 128: 1089-1095
        • Dahiya S.
        • Kumar R.
        • Sarkar C.
        • Ralte M.
        • Sharma M.C.
        Clear cell odontogenic carcinoma: a diagnostic dilemma.
        Pathol Oncol Res. 2002; 8: 283-285
        • Braunshtein E.
        • Vered M.
        • Taicher S.
        • Buchner A.
        Clear cell odontogenic carcinoma and clear cell ameloblastoma: a single clinicopathologic entity? A new case and comparative analysis of the literature.
        J Oral Maxillofac Surg. 2003; 61: 1004-1010
        • Kumar M.
        • Fasanmade A.
        • Barrett A.W.
        • Mack G.
        • Newman L.
        • Hyde N.C.
        Metastasising clear cell odontogenic carcinoma: a case report and review of the literature.
        Oral Oncol. 2003; 39: 190-194
        • Carinci F.
        • Volina S.
        • Rubini C.
        • Fioroni M.
        • Fracioso F.
        • Arcelli D.
        • Pezzetti F.
        • Piattelli A.
        Genetic profile of clear cell odontogenic carcinoma.
        J Craniofac Surg. 2003; 14: 356-362
        • Elbeshir E.L.
        • Harris M.
        • Barrett A.W.
        Clear cell odontogenic carcinoma of the maxilla: clinical, histological and immunohistochemical features of a case.
        Oral Oncol. 2004; 40: 91-94
        • Siriwardena B.S.
        • Tilakaratne W.M.
        • Rajapaksha R.M.S.K.
        Clear cell odontogenic carcinoma—a case report and review of literature.
        Int J Oral Maxillofac Surg. 2004; 33: 512-514
        • Ebert C.S.
        • Dubin M.G.
        • Hart C.F.
        • Chalian A.A.
        • Shockley W.W.
        Clear cell odontogenic carcinoma: a comprehensive analysis of treatment strategies.
        Head Neck. 2005; 27: 536-542
        • Avninder S.
        • Rakheja D.
        • Bhatnagar A.
        Clear cell odontogenic carcinoma: a diagnostic and therapeutic dilemma.
        World J Surg Oncol. 2006; 4: 91
        • Chera B.S.
        • Villaret D.B.
        • Orlando C.A.
        • Mendenhall W.M.
        Clear cell odontogenic carcinoma of the maxilla: a case report and literature review.
        Am J Otolaryngol. 2008; 29: 284-290
        • Xavier F.C.
        • Rodini C.O.
        • Rmalho L.M.P.
        • Sarmento V.A.
        • Nunes F.D.
        • de Sousa S.C.O.M.
        Clear cell odontogenic carcinoma: case report with immunohistochemical findings adding support to the challenging diagnosis.
        Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2008; 106: 403-410
        • Chaine A.
        • Pitak-Arnnop P.
        • Dhanuthai K.
        • Bertrand J.C.
        • Bertolus C.
        An asymptomatic radiolucent lesion of the maxilla. Clear cell odontogenic carcinoma.
        Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2009; 107: 452-457
        • Werle H.
        • Blake F.A.S.
        • Reichelt U.
        • Schmelzle R.
        • Heiland M.
        Clear-cell odontogenic carcinoma: a new case and long-term follow-up of an old case, and review of the literature.
        J Oral Maxillofac Surg. 2009; 67: 1342-1348
        • Zhang J.
        • Liu L.
        • Pan J.
        • Tian X.
        • Tan J.
        • Zhou J.
        • Duan Y.
        Clear cell odontogenic carcinoma: report of 6 cases and review of the literature.
        Med Oncol. 2011; 28: S626-S633
        • Barber B.
        • Cote D.
        • Seikaly H.
        Clearing up clear cell tumours of the head and neck: differentiation of hyalinizing and odontogenic varieties.
        J Otolaryngol Head Neck Surg. 2010; 39: E56-E60
        • Yazici Z.M.
        • Mete O.
        • Elmali Z.
        • Sayin I.
        • Yilmazer R.
        • Kayhan F.T.
        Clear cell odontogenic carcinoma of the maxilla.
        Acta Medica (Hradec Kralove). 2011; 54: 122-124
        • Coleman H.G.
        • Altini M.
        • Lurie R.
        Carcinomas of the jaws: report of a clear cell odontogenic carcinoma.
        Pathology. 2012; 44: S12
        • Dashow J.E.
        • McHugh J.B.
        • Edwards S.P.
        Swelling of the anterior mandible.
        J Oral Maxillofac Surg. 2012; 70: e204-e209
        • Alsheddi M.A.
        • Al Weieid A.
        • Al Shahrani F.
        • Al Faleh W.
        Clear cell odontogenic carcinoma or clear cell ameloblastoma: a case report and literature review.
        Oral Surg Oral Med Oral Pathol Oral Radiol. 2012; 114: E33
        • Khalam S.A.
        • Zachariah R.K.
        • Kumar S.
        Clear cell odontogenic carcinoma of mandible.
        Int J Dent Clin. 2012; 4: 55
        • Kenan S.
        • Ramazanoglu M.
        • Siraneci P.
        • Sofiyev N.
        • Olgac N.V.
        • Emekli U.
        • Onur O.D.
        Clear cell odontogenic carcinoma—a case report.
        Oral Oncol. 2013; 49: S106
        • Swain N.
        • Dhariwal R.
        • Ray J.G.
        Clear cell odontogenic carcinoma of maxilla: a case report and mini review.
        J Oral Maxillofac Pathol. 2013; 17: 89-94
        • Yancoskie A.E.
        • Sreekantaiah C.
        • Jabob J.
        • Rosenberg A.
        • Edelman M.
        • Antonescu C.R.
        • Fantasia J.E.
        EWSR1 and ATF1 rearrangements in clear cell odontogenic carcinoma: presentation of a case.
        Oral Surg Oral Med Oral Pathol Oral Radiol. 2014; 118: e115-e118
        • Devaraju R.R.
        • Reddy S.
        • Yashoda
        • Sekhar M.S.M.
        Clear cell odontogenic carcinoma versus hyalinizing clear cell carcinoma: a diagnostic dilemma.
        J Ind Acad Oral Med Radiol. 2014; 26: 319
        • Kalsi A.S.
        • Williams S.P.
        • Shah K.A.
        • Fasanmade A.
        Clear cell odontogenic carcinoma: a rare neoplasm of the maxillary bone.
        J Oral Maxillofac Surg. 2014; 72: 935-938
        • Krishnamurthy A.
        • Ramshankar V.
        • Majhi U.
        Clear cell odontogenic carcinoma of the mandible and temporomandibular joint with cervical lymph nodal metastasis.
        Natl J Maxillofac Surg. 2014; 5: 221-223
        • Kim M.
        • Cho E.
        • Kim J.Y.
        • Kim H.S.
        • Nam W.
        Clear cell odontogenic carcinoma mimicking a cystic lesion: a case of misdiagnosis.
        J Korean Assoc Oral Maxillofac Surg. 2014; 40: 199-203
        • Ghasemi Moridani S.
        • Edalat M.
        • Falahazad V.
        • Yazdanpanah S.
        Clear cell odontogenic carcinoma of mandible—a case report.
        J Res Dent Sci. 2014; 10: 10
        • Krishnamoorthy A.
        • Ravi Kumar A.S.
        • Batstone M.
        FDG-PET/CT in staging of clear cell odontogenic carcinoma.
        Int J Oral Maxillofac Surg. 2014; 43: 1326-1329
        • Ganvir S.M.
        • Gajbhiye N.Y.
        An unusual presentation of clear cell odontogenic carcinoma in mandibular anterior region.
        J Oral Maxillofac Pathol. 2014; 18: 442-448
        • Jahanbani J.
        • Ghasemi Moridani S.
        • Edalat M.
        Clear cell odontogenic carcinoma of mandible: a rare case report.
        Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2015; 119: e158
        • Harbhajanka A.
        • Lamzabi I.
        • Jain R.
        • Gattuso P.
        • Kluskens L.
        Cytomorphology and immunohistochemistry of a recurrent clear cell odontogenic carcinoma with molecular analysis: a case report with review of literature.
        Diagn Cytopathol. 2015; 43: 743-746
        • Prakash A.R.
        • Sairam V.
        • Srinivas Reddy P.
        Clear cell odontogenic carcinoma—a rare case report.
        J Maxillofac Oral Surg. 2015; 14: 60-63
        • Walia C.
        • Chatterjee R.P.
        • Kundu S.
        • Roy S.
        Clinical enigma: a rare case of clear cell odontogenic carcinoma.
        Contemp Clin Dent. 2015; 6: 559-563
        • Jain G.
        • Hegde P.
        • Shetty P.
        Clear cell odontogenic carcinoma: a rare case.
        Clin Cancer Investig J. 2015; 4: 408-410
        • Narula V.
        • Sharma D.
        • Bargava E.K.
        • Meher R.
        • Mandal S.
        • Rana K.
        Clear cell odontogenic carcinoma of maxilla: a rare case in a rarer presentation.
        J Oral Maxillofac Surg Med Pathol. 2016; 28: 95-99
        • Jayapalan C.S.
        • George A.
        • Noufal A.
        • Pynadath M.K.
        • Mangalath U.
        Clear cell odontogenic carcinoma (CCOC): mini-review of literature and case report of mandibular radiolucency in 17-year girl.
        Diagn Pathol Open. 2016; 1: 120
        • Ginat D.T.
        • Villafor V.
        • Cipriani N.A.
        Oral cavity clear cell odontogenic carcinoma.
        Head Neck Pathol. 2016; 10: 217-220
        • Rojas L.V.
        • Claudio C.A.
        • Pfuro V.R.
        An unusual presentation of clear cell odontogenic carcinoma: case report.
        Int J Dent Sci. 2016; 18: 33-39
        • Siraj F.
        • Kaur M.
        • Agrawal U.
        Clear cell odontogenic carcinoma of maxilla: a diagnostic challenge.
        Clin Cancer Investig J. 2016; 5: 256-258
        • Ordioni U.
        • Benat G.
        • Hadj Said M.
        • Gomez-Brouchet A.
        • Chossegros C.
        • Catherine J.H.
        Clear cell odontogenic carcinoma, diagnostic difficulties. A case report.
        J Stomatol Oral Maxillofac Surg. 2017; 118: 302-305
        • Datar U.V.
        • Kamat M.S.
        • Kanitkar S.S.
        • Byakodi S.S.
        Clear cell odontogenic carcinoma: a rare case report with emphasis on differential diagnosis.
        J Cancer Res Ther. 2017; 13: 374-377
        • Memtsa P.T.
        • Papadopoulou A.
        • Vachtsevanos K.
        • Tzitzikas I.
        Management of clear cell odontogenic carcinoma: a case report.
        J Med Cases. 2018; 9: 215-217
        • Kujiraoka S.
        • Tsunematsu T.
        • Sato Y.
        • Yoshida M.
        • Ishikawa A.
        • Tohyama R.
        • Tanaka M.
        • Kobayashi Y.
        • Kondo T.
        • Ushio A.
        • Otsuka K.
        • Kurosawa M.
        • Saito M.
        • Yamada A.
        • Arakaki R.
        • Nagai H.
        • Nikai H.
        • Takeuchi K.
        • Nagao T.
        • Miyamoto Y.
        • Ishimaru N.
        • Kudo Y.
        Establishment and characterization of a clear cell odontogenic carcinoma cell line with EWSR1–ATF1 fusion gene.
        Oral Oncol. 2017; 69: 46-55
        • Vogels R.
        • Baumhoer D.
        • van Gorp J.
        • Eijkelenboom A.
        • Verdijk M.
        • van Cleef P.
        • Bloemena E.
        • Slootweg P.J.
        • Lohman B.
        • Debiec-Rychter M.
        • Flucke U.
        Clear cell odontogenic carcinoma: occurrence of EWSR1–CREB1 as alternative fusion gene to EWSR1–ATF1.
        Head Neck Pathol. 2019; 13: 225-230