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Primordial Odontogenic Cyst with Induction Phenomenon (Zonal Fibroblastic Hypercellularity) and Dentinoid Material Versus Archegonous Cystic Odontoma: You Choose!

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Abstract

The most recent A.F.I.P. fascicle defines primordial odontogenic cyst (POC) as a distinct, nonkeratinized, odontogenic cyst of “undetermined origin” forming in the place of a developing normal or supernumerary tooth. However, the majority of examples reported in the literature under this term represent odontogenic keratocysts (keratocystic odontogenic tumors). In addition, there are rare reported cases of cystic odontomas. An 18-year-old Caucasian male presented with a unilocular mandibular radiolucent lesion in the place of a congenitally missing molar. Histologically, it featured nonkeratinizing, thin stratified squamous epithelial lining with areas of spongiosis and foci of vacuolization of individual basal cells without significant nuclear palisading. Focally, budding of the basal cell layer was identified. A zone of increased cellularity featuring induction-type fibroblasts was present subepithelially as well as dentinoid deposits with odontogenic epithelial nests. Immunohistochemically, the epithelial lining was negative for calretinin and the induction-like zone negative for S100 protein, smooth muscle actin, and CD34. The case was externally reviewed by five oral pathologists who provided various diagnostic interpretations including primordial cyst, odontogenic cyst not otherwise specified (NOS), cyst with ameloblastic changes, and unicystic ameloblastoma. At that time, a final diagnosis of odontogenic cyst NOS was rendered with a comment that it may represent a true example of POC or a cystic odontoma. The lesion has not recurred within a 13 year follow-up period after initial excision. An unusual cystic lesion is presented that may represent a true example of POC with dentinoid formation or an archegonous cystic odontoma.

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References

  1. Vickers RA, Gorlin RJ. Ameloblastoma: delineation of early histopathologic features of neoplasia. Cancer. 1970;26:699–710.

    Article  CAS  PubMed  Google Scholar 

  2. Altini M, Coleman H, Doglioni C, Favia G, Maiorano E. Calretinin expression in ameloblastomas. Histopathology. 2000;37:27–32.

    Article  CAS  PubMed  Google Scholar 

  3. Coleman H, Altini M, Ali H, Doglioni C, Favia G, Maiorano E. Use of calretinin in the differential diagnosis of unicystic ameloblastomas. Histopathology. 2001;38:312–7.

    Article  CAS  PubMed  Google Scholar 

  4. DeVilliers P, Liu H, Suggs C, Simmons D, Daly B, Zhang S, Raubenheimer E, Larsson A, Wright T. Calretinin expression in the differential diagnosis of human ameloblastoma and keratocystic odontogenic tumor. Am J Surg Pathol. 2008;32:256–60.

    Article  PubMed  Google Scholar 

  5. Anandani C, Metgud R, Singh K. Calretinin as a diagnostic adjunct for ameloblastoma. Pathol Res Int. 2014;2014:308240.

    Article  Google Scholar 

  6. Robinson HBG. Classification of cysts of the jaws. Am J Orthod Oral Surg. 1945;31:370–5.

    Article  Google Scholar 

  7. Robinson HB. Primordial cyst versus keratocyst. Oral Surg Oral Med Oral Pathol. 1975;40:362–4.

    Article  CAS  PubMed  Google Scholar 

  8. Robinson HBG, Koch WE Jr, Kolas S. Radiographic Interpretation of oral cysts. Dent Radiogr Photogr. 1956;29:61–8.

    Google Scholar 

  9. Philipsen HP. Om keratocystes (kolesteatomer) i kaeberne. Tandlaegebl. 1956;69:963–80.

    Google Scholar 

  10. Partridge M, Towers JF. The primordial cyst (odontogenic keratocyst): its tumour-like characteristics and behaviour. Br J Oral Maxillofac Surg. 1987;25:271–9.

    Article  CAS  PubMed  Google Scholar 

  11. Emerson TG, Whitlock RI, Jones JH. Involvement of soft tissue by odontogenic keratocysts (primordial cysts). Br J Oral Surg. 1972;9:181–5.

    Article  CAS  PubMed  Google Scholar 

  12. Shear M, Altini M. Odontogenic and non-odontogenic cysts of the jaws. J Dent Assoc S Afr. 1983;38:555–60.

    CAS  PubMed  Google Scholar 

  13. Pindborg JJ, Kramer IRH. WHO histological typing of odontogenic tumors. Geneva: Jaw Cysts and Allied Lesions; 1971.

    Google Scholar 

  14. Philipsen HP. Keratocystic odontogenic tumour. In: Barnes L, Eveson JW, Reichart P, Sidransky D, editors. WHO classification of tumours. Pathology and genetics of head and neck tumours. Lyon: IARC Press; 2005. p. 306–7.

    Google Scholar 

  15. Robinson RA, Vincent SD. Primordial cyst (odontogenic cyst of undetermined origin). In: Silverberg SG, editor. AFIP Atlas of tumor pathology, fourth series, Fascicle 16, tumors and cysts of the jaws. Silver Spring, Maryland: ARP Press; 2012.

    Google Scholar 

  16. Brannon RB. The odontogenic keratocyst. A clinicopathologic study of 312 cases. Part I. Clinical features. Oral Surg Oral Med Oral Pathol. 1976;42:54–72.

    Article  CAS  PubMed  Google Scholar 

  17. Bernier JL. Simple follicular (primordial) cyst. In: AFIP Atlas of tumor pathology, section IV, Fascicle 10a, tumors of the odontogenic apparatus and jaws. Washington: ARP Press; 1960.

  18. Hoffman S, Jacoway JR, Krolls SO. Primordial cyst. In: Hoffman S, editor. AFIP Atlas of tumor pathology, second series, Fascicle 24, intraosseous and parosteal tumors of the jaws. Washington: ARP Press; 1987.

    Google Scholar 

  19. Sciubba JJ, Fantasia JE, Kahn LB. Primordial cyst. In: Rosai J, editor. AFIP Atlas of tumor pathology, third series, Fascicle 29, tumors and cysts of the jaws. Washington: ARP Press; 2001.

    Google Scholar 

  20. Browne RM. The pathogenesis of the odontogenic keratocyst. In: Fourth proceedings of the international academy of oral pathology; 1969. p. 28.

  21. Forssell K. The primordial cyst. A clinical and radiographic study. Proc Finn Dent Soc. 1980;76:129–74.

    CAS  PubMed  Google Scholar 

  22. Reff-Eberwein G, Donath K, Schmitz R. Odontogenic keratocysts (OKC). Histologic and clinical follow-up studies. Dtsch Zahnarztl Z. 1985;40:514–20 (passim).

    CAS  PubMed  Google Scholar 

  23. Mistry D, Altini M, Coleman HG, Ali H, Maiorano E. The spatial and temporal expression of calretinin in developing rat molars (Rattus norvegicus). Arch Oral Biol. 2001;46:973–81.

    Article  CAS  PubMed  Google Scholar 

  24. Koneru A, Hallikeri K, Nellithady GS, Krishnapillai R, Prabhu S. Immunohistochemical expression of calretinin in ameloblastoma, adenomatoid odontogenic tumor, and keratocystic odontogenic tumor: a comparative study. Appl Immunohistochem Mol Morphol. 2014;22:762–7.

    Article  CAS  PubMed  Google Scholar 

  25. Usubütün A, Atayar C, Basal N, Araz K. Cystic ameloblastic fibroma. Br J Oral Maxillofac Surg. 2002;40:512–4.

    Article  PubMed  Google Scholar 

  26. Ng KH, Siar CH. Odontogenic keratocyst with dentinoid formation. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2003;95:601–6 (Review).

    Article  PubMed  Google Scholar 

  27. Slabbert H, Altini M, Crooks J, Uys P. Ameloblastoma with dentinoid induction: dentinoameloblastoma. J Oral Pathol Med. 1992;21:46–8.

    Article  CAS  PubMed  Google Scholar 

  28. Matsumoto Y, Mizoue K, Seto K. Atypical plexiform ameloblastoma with dentinoid: adenoid ameloblastoma with dentinoid. J Oral Pathol Med. 2001;30:251–4 (Review).

    Article  CAS  PubMed  Google Scholar 

  29. Evans BL, Carr RF, Phillipe LJ. Adenoid ameloblastoma with dentinoid: a case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2004;98:583–8.

    Article  PubMed  Google Scholar 

  30. Shafer WG, Hine MK, Levy BM, editors. A textbook of oral pathology. 2nd ed. Philadelphia: W.B. Saunders; 1963. p. 209–10.

    Google Scholar 

  31. Ide F, Obara K, Mishima K, Saito I, Horie N, Shimoyama T, Kusama K. Peripheral odontogenic tumor: a clinicopathologic study of 30 cases. General features and hamartomatous lesions. J Oral Pathol Med. 2005;34:552–7.

    Article  CAS  PubMed  Google Scholar 

  32. Goldberg H, Schofield ID, Popowich LD, Wakeham D. Cystic complex composite odontoma. Report of two cases. Oral Surg Oral Med Oral Pathol. 1981;51:16–20.

    Article  CAS  PubMed  Google Scholar 

  33. Thoma KH. Tumors of odontogenic origin. In: Oral pathology. A histological, roentgenological, and clinical study of the diseases of the teeth, jaws, and mouth. St. Louis: C.V. Mosby; 1941. p. 960–8.

  34. Mosqueda-Taylor A, Pires FR, Aguirre-Urízar JM, et al. Primordial odontogenic tumour: clinicopathological analysis of six cases of a previously undescribed entity. Histopathology. 2014;65:606–12.

    Article  PubMed  Google Scholar 

  35. Ide F, Kikuchi K, Kusama K, Muramatsu T. Primordial odontogenic tumour: is it truly novel? Histopathology. 2015;66:603–4.

    Article  PubMed  Google Scholar 

  36. Morita S, Jogetsu K, Nosaka Y, et al. A case of odontogenic fibroma in the mandible of a three-year-old boy (Japanese with English abstract). Jpn J Oral Maxillofac Surg. 1994;40:935–7.

    Article  Google Scholar 

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Acknowledgments

The authors are grateful to Drs. Carl Allen, Paul Freedman, Raymond Melrose, Mervyn Shear and Lee Slater for providing their expert opinion as consultants. Finally, we are indebted to Mr. Brian Dunnette (University of Minnesota) for his assistance with the illustrations.

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Authors and Affiliations

  1. Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, 515 Delaware Street SE 16-206B, Minneapolis, MN, 55455, USA

    Prokopios P. Argyris, Stephanie L. Wetzel, Rajaram Gopalakrishnan & Ioannis G. Koutlas

  2. Department of Laboratory Medicine and Pathology, University of Minnesota Medical Center, University of Minnesota, Minneapolis, MN, USA

    Stefan E. Pambuccian

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  1. Prokopios P. Argyris
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  2. Stephanie L. Wetzel
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Correspondence to Ioannis G. Koutlas.

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Argyris, P.P., Wetzel, S.L., Pambuccian, S.E. et al. Primordial Odontogenic Cyst with Induction Phenomenon (Zonal Fibroblastic Hypercellularity) and Dentinoid Material Versus Archegonous Cystic Odontoma: You Choose!. Head and Neck Pathol 10, 237–244 (2016). https://doi.org/10.1007/s12105-015-0640-2

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