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Calcifying Epithelial Odontogenic Tumour with Clear Langerhans Cells: A Novel Variant, Report of a Case and Review of the Literature

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Abstract

Clear cell calcifying epithelial odontogenic tumour (CCEOT) is a rare variant of calcifying epithelial odontogenic tumor (CEOT). While it is not surprising to find clear cells in odontogenic lesions, the exact nature of the clear cells in CCEOT has not been elucidated. Herein, we report a case of peripheral CCEOT of anterior mandible in a 37 year old black female. Histologically, the tumour consisted of cords and small nests of clear cells surrounded by dense deposits of amyloid and basophilic calcifications. The cells possessed abundant clear cytoplasm and eccentrically located indented nuclei. Admixed with the clear cells were eosinophilic cuboidal to polyhedral cells. The clear cells were PAS negative and immunoreactive for S100 protein, CD1a and Langerin. The clear cells were negative for MNF-116, SMA, Desmin and CK-19. It is therefore recommended to recognize two variants of CCEOT, namely, CEOT with clear cell change and CEOT with clear Langerhans cells (LC). We further suggest that the contradictory term “non-calcifying variant of calcifying epithelial odontogenic tumour with LC” to be abandoned, as the current case clearly indicates that LC could be seen in CEOT irrespective of the presence or absence of calcifications.

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References

  1. Asano M, Takahashi T, Kusama K, Iwase T, Hori M, Yamanoi H, Tanaka H, Maro I. A variant of calcifying epithelial odontogenic tumour with Langerhans cells. J Oral Pathol Med. 1990;19:430–4.

    Article  CAS  PubMed  Google Scholar 

  2. Takata T, Ogawa I, Miyauchi M, Ijuhin N, Nikai H, Fujita M. Non-Calcifying Pindborg tumour with Langerhans cells. J Oral Pathol Med. 1993;22:378–83.

    Article  CAS  PubMed  Google Scholar 

  3. Li Wang, Wang S, Chen X. Langerhans cells containing calcifying epithelial odontogenic tumour: Report of two cases and review of the literature. Oral Oncol Extra. 2006;42:144–6.

    Article  Google Scholar 

  4. Wang YP, Wang JT, Liu BY, Yu CH, Kuo RC, Chiang CP. Non-calcifying variant of calcifying epithelial odontogenic tumour with Langerhans cells. J Oral Pathol Med. 2007;36:436–9.

    Article  CAS  PubMed  Google Scholar 

  5. Pindborg JJ. A calcifying epithelial odontogenic tumor. Cancer. 1958;11:838–43.

    Article  CAS  PubMed  Google Scholar 

  6. Abrams AM, Howell FV. Calcifying epithelial odontogenic tumours: report of four cases. J Am Dent Assoc. 1967;74:1231–40.

    CAS  PubMed  Google Scholar 

  7. Rangel ALCA, da Silva AA, Ito FA, Lopes MA, de Almeida OP, Vargas PA. Clear cell variant of calcifying epithelial odontogenic tumour: is it locally aggressive? J Oral Maxillofac Surg. 2009;67:207–11.

    Article  PubMed  Google Scholar 

  8. Anavi Y, Kaplan I, Citir M, Calderon S. Clear-cell variant of calcifying epithelial odontogenic tumour: clinical and radiographic characteristics. Oral Surg Oral Med Oral Pathol. 2003;95:332–9.

    Article  Google Scholar 

  9. Philipsen HP, Reichart PA. Calcifying epithelial odontogenic tumour: biological profile based on 181 cases from the literature. Oral Oncol. 2000;36:17–26.

    Article  CAS  PubMed  Google Scholar 

  10. de Augiar MCF, Gomez RS, Silva EC, de Araujo VC. Clear cell ameloblastoma (clear cell odontogenic carcinoma): report of a case. Oral Surg Oral Med Oral Pathol. 1996;81:79–83.

    Article  Google Scholar 

  11. de Oliveira MG, Chaves ACM, Visioli F, Rojas EU, Moure SP, Romanini J, ana Filho MS. Peripheral clear cell variant of calcifying epithelial odontogenic tumour affecting 2 sites: report of a case. Oral Surg Oral Med Oral Pathol. 2009;107:407–11.

    Article  Google Scholar 

  12. Houston GD, Fowler CB. Extraosseous calcifying epithelial odontogenic tumour. Report of two cases and review of the literature. Oral Surg Oral Med Oral Pathol. 1997;83:577–83.

    Article  CAS  Google Scholar 

  13. Haustein UF. The Langerhans cell–its macrophages-analogous function in the triggering of the allergic contact eczema (author’s transl). Allerg Immunol (Leipz). 1979;25:116–31.

    CAS  Google Scholar 

  14. Cocchia D, Michetti F, Donato R. Immunochemical and immuno-cytochemical localization of S-100 antigen in normal human skin. Nature. 1981;294:85–7.

    Article  CAS  PubMed  Google Scholar 

  15. Nakajima T, Kodama T, Tsumuraya M, Shimosato Y, Kameya T. S-100 protein-positive Langerhans cells in various human lung cancers, especially in peripheral adenocarcinomas. Virchows Arch A Pathol Anat Histopathol. 1985;407:177–89.

    Article  CAS  PubMed  Google Scholar 

  16. Ambe K, Mori M, Enjoji M. S-100 protein-positive dendritic cells in colorectal adenocarcinomas. Distribution and relation to the clinical prognosis. Cancer. 1989;63:496–503.

    Article  CAS  PubMed  Google Scholar 

  17. Nomori H, Watanabe S, Nakajima T, Shimosato Y, Kameya T. Histiocytes in nasopharyngeal carcinoma in relation to prognosis. Cancer. 1986;57:100–5.

    Article  CAS  PubMed  Google Scholar 

  18. Azizi E, Bucana C, Goldberg L, Kripke ML. Perturbation of epidermal Langerhans cells in basal cell carcinomas. Am J Dermatopathol. 1987;6:465–73.

    Article  Google Scholar 

  19. O’Donnell RK, Mick R, Feldman M, Hino S, Wang Y, Brose MS, Muschel RJ. Distribution of dendritic cell subtypes in primary oral squamous cell carcinoma is inconsistent with a functional response. Cancer Lett. 2007;255:145–52.

    Article  PubMed Central  PubMed  Google Scholar 

  20. McCarter MD, Baumgartner J, Escobar GA, Richter D, Lewis K, Robinson W, Wilson C, Palmer BE, Gonzalez R. Immunosuppressive dendritic and regulatory T cells are upregulated in melanoma patients. Ann Surg Oncol. 2007;14:2854–60.

    Article  PubMed  Google Scholar 

  21. Gatter KC, Morris HB, Roach B, Mortimer P, Fleming KA, Mason DY. Langerhans’ cells and T cells in human skin tumours: an immunohistological study. Histopathology. 1984;8:229–44.

    Article  CAS  PubMed  Google Scholar 

  22. Kumar M, Fasanmade A, Barrett AW, Mack G, Newman L, Hyde NC. Metastasizing clear cell odontogenic carcinoma: a case report and review of the literature. Oral Oncol. 2003;39:190–4.

    Article  PubMed  Google Scholar 

  23. Kauzman A, Tabet JC, Stiharu TI. Hyalinizing clear cell carcinoma: a case report and review of the literature. Oral Surg Oral Med Oral Pathol. 2011;112:e26–34.

    Article  Google Scholar 

  24. Bilodeau EA, Weinreb I, Antonescu CR, Zhang L, Dacic S, Muller S, Barker B, Seethala 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;37:1001–5.

    Article  PubMed  Google Scholar 

  25. Okabe M, Miyabe S, Nagatsuka H, Terada A, Hanai N, Yokoi M, Shimozato K, Eimoto T, Nakamura S, Nagai N, Hasegawa Y, Inagaki H. MECT1-MAML2 fusion transcript defines a favorable subset of mucoepidermoid carcinoma. Clin Cancer Res. 2006;12:3902–7.

    Article  CAS  PubMed  Google Scholar 

  26. Hicks MJ, Flaitz CM, Wong ME, McDaniel RK, Cagle PT. Clear cell variant of calcifying epithelial odontogenic tumor: case report and review of the literature. Head Neck. 1994;16:272–7.

    Article  CAS  PubMed  Google Scholar 

  27. Hansen LS, Eversole LR, Green TL, Powell NB. Clear cell odontogenic tumour: a new entity with aggressive potential. Head Neck Surg. 1985;8:115–23.

    Article  CAS  PubMed  Google Scholar 

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Acknowledgments

We would like to thank Dr. Ismail Gopal, head of oral and maxillofacial surgery unit, Livingstone hospital, for providing us with the radiograph, Mr. Wessel Kleinhans, IT specialist, NHLS, for his technical support, Mrs. Claudette Gobregts, consultant sales manager, SMM instruments, for providing the antibody Langerin and finally Mr. Ben Henrico, head of immunohistochemistry, Tygerberg Laboratories, for his kind assistance.

Conflict of interest

The authors declare that they have no conflict of interest.

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Authors and Affiliations

  1. Department of Oral Pathology, NHLS, Tygerberg Hospital, University of the Western Cape, Cape Town, South Africa

    Amir Afrogheh & Jos Hille

  2. Division of Anatomical Pathology, NHLS, Tygerberg Hospital, University of Stellenbosch, Cape Town, South Africa

    Johann Schneider & Noor Mohamed

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  1. Amir Afrogheh
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  2. Johann Schneider
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  3. Noor Mohamed
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  4. Jos Hille
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Correspondence to Amir Afrogheh.

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Afrogheh, A., Schneider, J., Mohamed, N. et al. Calcifying Epithelial Odontogenic Tumour with Clear Langerhans Cells: A Novel Variant, Report of a Case and Review of the Literature. Head and Neck Pathol 8, 214–219 (2014). https://doi.org/10.1007/s12105-013-0490-8

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  • DOI: https://doi.org/10.1007/s12105-013-0490-8

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