‘Hybrid’ desmoplastic ameloblastoma: an unusual case report with immunohistochemical investigation for TGF-β and review of literature

Abstract

Abstract. Ameloblastoma, a relatively common epithelial odontogenic tumor includes several histopathologic subtypes like follicular, plexiform, acanthomatous and desmoplastic variants. Hybrid desmoplastic ameloblastoma (DA) composed of typical desmoplastic ameloblastoma along with areas of follicular/plexiform ameloblastoma is an extremely rare variant of ameloblastoma and to date only 16 cases have been documented. We report a new case of hybrid DA which additionally showed remarkable histologic findings of extensive bone formation and presence of mucous cells. It occurred in a 64 year old female patient involving the left maxilla from lateral incisor to distal to first molar. A comprehensive review of hybrid DA and the unusual findings aimed to offer an insight into the pathogenesis of these heterogeneities in odontogenic epithelium and clinical implications was also undertaken. Hybrid DA showed characteristic clinicopathologic features of a desmoplastic ameloblastoma with the exception of predominant mandibular occurrence. A likelihood of areas of solid multicystic ameloblastoma being a part of histopathologic spectrum of DA questioning the need for use of term “hybrid” was observed. Regarding the osteoplasia, most cases occurred in Asians and in anterior maxilla. The prominent TGF-β expression seen in our case adds emphasis to its role in desmoplasia and osteoplasia. Furthermore, we identified an interesting association of desmoplasia with incidence of mucous cell metaplasia in ameloblastoma.

 

Key words: Ameloblastoma; hybrid desmoplastic ameloblastoma, osteoplasia, mucous cells, TGF-β

Keywords

• Ameloblastoma, hybrid desmoplastic ameloblastoma, osteoplasia, mucous cells, TGF-β

Kaynakça

1. Reichart PA, Philipsen HP. Odontogenic tumors and allied lesions. Quintessesnce Publishing Co. London p69-p77, 2004, 1 st edition.
2. Reichart PA, Philipsen HP, Sonner S. Ameloblastoma: biological profile of 3677 cases. Eur J Cancer B Oral Oncol 1995; 31: 86–99.
3. Shashikant MC, Neetha MC, Ali IM, Shambulingappa P. Desmoplastic ameloblastoma in the maxilla: a case report and review of literature. Indian J Dent Res 2007; 18: 214-217.
4. Philipsen HP, Reichart PA, Takata T. Desmoplastic ameloblastoma (including “hybrid” lesion of ameloblastoma. Biological profile based on 100 cases from literature and own files. Oral Oncol 2001; 37: 455-460.
5. Eversole LR, Leider AS, Hansen LJ. Ameloblastomas with pronounced desmoplasia.J Oral Maxillofac Surg 1984; 42: 735-740.
6. Kishino M, Murakama S, Fukuda Y, Ishida T. Pathology of desmoplastic ameloblastoma. J Oral Pathol Med 2001; 30: 35-40.
7. Beckley ML, Farhood V, Helfend LK, Alijanian A. Desmoplastic ameloblastoma of the mandible: a case report and review of literature. J Oral Maxillofac Surg 2002; 60: 194-198.
8. Sun ZJ, Wu YR, Cheng N, Zwahlen RA, Zhao YF. Desmoplastic Ameloblastoma-A review. Oral Oncol 2009; 45: 742-759.

9. Sakashita H, Miyata M, Okabe K, Kurumaya H. Desmoplastic ameloblastoma in the maxilla: a case report. J Oral Maxillofac Surg 1998; 56: 783-786.
10. Gardner DG, Heikinheimo K, Shear M, Philipsen H P, Coleman H. Ameloblastomas. In: L. Barnes, J.W. Eveson, P Reichart and D. Sidransky, (Eds). World health organization classification of tumors. Pathology and genetics of head and neck tumors, IARC Press, Lyon; 2005. pp.296-300.
11. Waldron CA, El-Mofty SK. A histopathologic study of 116 ameloblastomas with special reference to desmoplastic variant. . Oral Surg Oral Med Oral Pathol 1987; 63: 441-451.
12. Philipsen HP, Ormiston IW, Reichart PA. The desmo-and osteoplastic ameloblastoma. Histologic variant or clinicopathologic entity? Case reports. J Oral Maxillofac Surg 1992; 21: 352-357.
13. Okada Y, Sugimura M, Ishida T. Ameloblastoma accompanied by prominent bone formation. J Oral Maxillofac Surg 1986; 555-557.
14. Thompson IOC, Van Rensburg LJ, Phillips VMJ. Desmoplastic ameloblastoma: correlative histopathology, radiology and CT-MR imaging. J Oral Pathol Med 1996; 25: 405-410.
15. Iida S, Kogo M, Kishino M, Matsuya T. Desmoplastic ameloblastoma with large cystic change in the maxillary sinus: report of a case. J Oral Maxillofac Surg 2002; 60: 1195-1198.
16. Angadi PV, Rekha K. “Ameloblastoma with mucous cells” : Review of literature and presentation of 2 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2008; 106: e20-e26.
17. Kawai T, Kishino M, Hiranuma H, Tadashi S, Ishida T. A unique case of desmoplastic ameloblastoma of the mandible; report of a case and brief review of English language literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1999; 87: 258-263.
18. Beckley ML, Farhood V, Helfend LK, Alijanian A. Desmoplastic ameloblastoma of the mandible : a case report and review of literature. J Oral Maxillofac Surg 2002; 60: 194-198.
19. Pillai RS, Ongole R, Ahsan A, Radhakrishnan RA, Pai KM. Recurrent desmoplastic ameloblastoma of the maxilla: a case report. J Can Dent Assoc 2004; 70: 100-104.
20. Takata T, Miyauchi M, Ito H, et al. Clinical and histopathological analysis of desmoplastic ameloblastoma. Pathol Res Pract 1999; 195: 669- 675.
21. Higuchi Y, Nakamura N, Ohishi M, Tashiro H. Unusual ameloblastoma with extensive stromal desmoplasia. J Cranio –Max Fac Surg 1991; 19: 323- 327.
22. Philipsen HP, Ormiston IW, Reichart PA. The desmo-and osteoplastic ameloblastoma. Histologic variant or clinicopathologic entity? Case reports. J Oral Maxillofac Surg 1992; 21: 352-357.
23. Ashman SG, Corio LC, Eisele DW, Murphy MT. Desmoplastic ameloblastoma: a case report and literature review. Oral Surg Oral Med Oral Pathol 1993; 75: 479-482.
24. Takata T, Miyauchi M, Ogawa I, et al. So-called ‘hybrid’ lesion of desmoplastic and conventional ameloblastoma: Report of a case and review of literature. Pathol Int 1999; 49: 1014-1018.
25. Wakoh M, harada T, Inoue T. Follicular/desmoplastic hybrid ameloblastoma with radiographic features of concomitant fibro-osseous and solitary cystic lesions. . Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002; 94: 774-780.
26. Hirota M, Aoki S, Kawabe R, Fujita K. Desmoplastic ameloblastoma featuring basal cell ameloblastoma :a case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2005; 99: 160-164.
27. Santos JND, De Souza VF, Azevedo RA, Sarmento VA, Souza LB.“Hybrid” lesion of desmoplastic and conventional ameloblastoma: immuno-histochemical aspects. Rev Bras Otorrinolaringol 2006; 72: 709- 713.
28. Sivapatha sundaram B, Einstein A, Syed RI. Desmoplastic ameloblastoma in Indians: Report of five cases and review of literature. Indian J Dent Res 2007; 18: 218-221.
29. Yazdi I, Seyedmajidi M, Foroughi R. Desmoplastic ameloblastoma(a hybrid variant):report of a case and review of the literature. Arch Iranian Med 2009; 12: 304-308.
30. Melrose RJ. Desmoplastic Ameloblastoma. Pathology Review 1999; 195: 669-675.
31. Raubenheimer EJ, van Heerden WF, Noffke CE. Infrequent clinicopathological findings in 108 ameloblastomas. J Oral Pathol Med 1995; 24: 227- 232.
32. Nakamura N, Higuchi Y, Tashiro H. Clinical and histopathological studies of ameloblastoma: ameloblastoma in the alveolar region. Japanese Journal of Oral Maxillofacial surgery 1986; 32: 829- 832 (In Japanese)
33. Takemoto T, Yamashita T, Ito A. A case of ameloblastoma of maxilla with bone tissue in the tumor. Japanese Journal of Oral Maxillofacial surgery 1991; 37: 234-239 (In Japanese)
34. Ishigami T, Sugihara K, Uchiyama T. Ameloblastoma of the maxilla with bone tissue in the tumor:report of a case. Japanese Journal of Oral Maxillofacial surgery 1991; 37: 2103-2104 (In Japanese)
35. Takata T, Miyauchi M, Ogawa I, et al. Immunoexpression of transforming growth factor β in desmoplastic ameloblastoma. Virchows Arch 2000; 436: 319-323.
36. Noda M, Camillerie JJ. In vivo stimulation of bone formation by transforming growth factor beta. Endocrinology 1989; 124: 2991-2994.
37. Centrella M, McCarthy TL, Canalis E. Transforming growth factor beta is a bifunctional regulator of replication and collagen synthesis in osteoblast enriched cell cultures from fetal rat bone J Biol Chem 1987; 262: 2869-2874.
38. Wrana JL, Maeono M, Hawrylyshyn B, et al. Differential effects of transforming growth factor beta on the synthesis of extracellular matrix proteins by normal fetal rat calvarial one populations. J Cell Biol 1988; 106: 915-924.
39. Bumpous JM. Metaplastic bone formation in nasal polyps with histologic presence of transforming growth factor –β1 and bone morphogenic proteins. Otolaryngology-Head and Neck Surgery 2001; 125: 96-97.
40. Iqbal J, Sun L, Zaidi M. Coupling bone degradation to bone formation. Nat Med 2009; 15: 729-731.
41. Dardick I, Birek C, Lingen MW, Rowe PE. Differentiation and cytomorphology of salivary gland tumors with specific reference to oncocytic metaplasia. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1999; 88: 691-701.
42. Wilson D, Walker M, Aurora N, Moore S. Ameloblastoma with mucous cell differentiation. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2001; 91: 576-578.
43. Hodson JJ. Observations on the origin and nature of the adamantinoma with special reference to certain muco-epidermoid variations. Br J Plast Surg 1957; 10: 38-59.
44. Browne RM. Metaplasia and degeneration in odontogenic cysts in man. J. Oral Path 1972; 1: 145- 158.
45. Fujiwara K, Watanabe T. Mucous-producing cells and ciliated epithelial cells in mandibular radicular cyst: an electron microscopic study. J Oral Maxillofac Surg 1988; 46: 149-151.
46. Gorlin RJ. Potentialities of oral epithelium manifest by mandibular dentigerous cysts. Oral Surg Oral Med Oral Pathol 1957; 10: 271.
47. Taxy JB. Necrotizing squamous /mucinous metaplasia in oncocytic salivary gland tumors. A potential diagnostic problem. Am J Clin Pathol 1992; 97: 40-45.
48. Kezler A, Paparella ML, Dominguez FV. Desmoplastic and non-desmoplastic ameloblastoma : a comparative clinicopathological analysis. Oral Dis 1996; 2: 228-231.