Imaging Sci Dent.  2013 Sep;43(3):201-207. 10.5624/isd.2013.43.3.201.

A repeatedly recurrent desmoplastic ameloblastoma after removal and allobone graft: Radiographic features compared with histological changes

• Kim JD
• Jang HS
• Seo YS
• Kim JS

• KMID: 2167459
• DOI: http://doi.org/10.5624/isd.2013.43.3.201

Abstract

A 40-year-old man suffered from a repeatedly recurrent desmoplastic ameloblastoma in the right maxillary anterior and premolar regions. During the first visit, the patient was provisionally histopathologically diagnosed with a developmental cyst, and it was confirmed to be unicystic ameloblastoma and resected. Four years later, the lesion recurred, and was diagnosed as a desmoplastic type of ameloblastoma and removed again. Then, 5 years after the second surgery, the lesion recurred again, and was diagnosed as a type containing a follicular pattern, recurrent ameloblastoma. A panoramic radiograph showed a multilocular and mixed radiolucent/radiopaque expansile lesion at the first visit, a unilocular cystic lesion confined to the premolar area at the second visit, and a small soap bubble appearance in the molar area in the final visit. Cone-beam computed tomographic images of the final recurrence of the tumor revealed multiple small cyst-like structures in the right maxillary anterior and posterior regions.

Keyword

Ameloblastoma; Recurrence; Cone-Beam Computed Tomography; Radiography, Panoramic

MeSH Terms

Adult
Ameloblastoma
Bicuspid
Cone-Beam Computed Tomography
Humans
Molar
Radiography, Panoramic
Recurrence
Soaps
Soaps

Figure

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  Fig. 1 Panoramic radiograph reveals a multilocular and mixed radiolucent/radiopaque expansile lesion extending from the right second premolar to the left canine of the maxilla, showing a soap bubble appearance in the middle third of the lesion, and an ill-defined radiopacity in the left third of the lesion.

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  Fig. 2 A. The incisional biopsy shows a cystic structure lined by epithelium (H&E stain, 200×). B. The histopathologic examination after the first surgery on the tumor shows peripheral palisading of hyperchromatic epithelium and loose fibrous stroma (H&E stain, 200×).

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  Fig. 3 A panoramic radiograph reveals a unilocular radiolucent cystic area confined to the premolar region of the right maxilla, 4 years after the first surgery.

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  Fig. 4 A. Histopathologic examination 4 years after the first surgery shows the desmoplastic type of ameloblastoma (H&E stain, 40×). B. The histopathologic features contain small islands and thin cords of ameloblastic epithelium within a dense fibrous connective tissue stroma (H&E stain, 200×).

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  Fig. 5 Five years after the second surgery. The panoramic radiograph reveals a soap bubble appearance in the periapical area of the right first and second molars of the maxilla.

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  Fig. 6 An axial view of the CBCT image shows multiple small cyst-like structures in the right anterior and posterior regions. They are separated by normal bone. The lesion in the molar region is well demarcated, and separated from the maxillary antrum.

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  Fig. 7 Sagittal CT images (A; molar area B; premolar area C: anterior area) show small loculations in the molar and anterior region of the maxilla, respectively, separated by normal bone. The tumor shows a relatively defined border, and it is adjacent to the maxillary sinus.

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  Fig. 8 A. The histopathologic examination 5 years after the second surgery reveals small cystic tumor islands and thin cords of ameloblastic epithelium within connective tissue stroma (H&E stain, 40×). B. A follicular pattern next to the desmoplastic type of ameloblastoma (H&E stain, 200×).

Reference

1. White SC, Pharoah MJ. Oral radiology: principles and interpretation. 6th ed. St. Louis: Mosby Elsevier;2009. p. 373–375.

2. Becelli R, Carboni A, Cerulli G, Perugini M, Iannetti G. Mandibular ameloblastoma: analysis of surgical treatment carried out in 60 patients between 1977 and 1998. J Craniofac Surg. 2002; 13:395–400.

3. Hertog D, Schulten EA, Leemans CR, Winters HA, Van der Waal I. Management of recurrent ameloblastoma of the jaws: a 40-year single institution experience. Oral Oncol. 2011; 47:145–146.

4. Rapidis AD, Andressakis DD, Stavrianos SD, Faratzis G, Arnogiannaki-Liappi N, Lagogiannis GA, et al. Ameloblastomas of the jaws: clinico-pathological review of 11 patients. Eur J Surg Oncol. 2004; 30:998–1002.

5. Siar CH, Lau SH, Ng KH. Ameloblastoma of the jaws: a retrospective analysis of 340 cases in a Malaysian population. J Oral Maxillofac Surg. 2012; 70:608–615.

6. Dissanayake RK, Jayasooriya PR, Siriwardena DJ, Tilakaratne WM. Review of metastasizing (malignant) ameloblastoma (METAM): pattern of metastasis and treatment. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2011; 111:734–741.

7. Mendenhall WM, Werning JW, Fernandes R, Malyapa RS, Mendenhall NP. Ameloblastoma. Am J Clin Oncol. 2007; 30:645–648.

8. Ladeinde AL, Ogunlewe MO, Bamgbose BO. Ameloblastoma: analysis of 207 cases in a Nigerian teaching hospital. Quintessence Int. 2006; 37:69–74.

9. Zemann W, Feichtinger M, Kowatsch E, Kärcher H. Extensive ameloblastoma of the jaws: surgical management and immediate reconstruction using microvascular flaps. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2007; 103:190–196.

10. Reichart PA, Philipsen HP, Sonner S. Ameloblastoma: biological profile of 3677 cases. Eur J Cancer B Oral Oncol. 1995; 31B:86–99.

11. Kim SG, Jang HS. Ameloblastoma: a clinical, radiographic, and histopathologic analysis of 71 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2001; 91:649–653.

12. Waldron CA, El-mofty SK. A histopathologic study of 116 ameloblastomas with special reference to the desmoplastic variant. Oral Surg Oral Med Oral Pathol. 1987; 63:441–451.

13. Kaffe I, Buchner A, Taicher S. Radiologic features of desmoplastic variant of ameloblastoma. Oral Surg Oral Med Oral Pathol. 1993; 76:525–529.

14. Ng KH, Siar CH. Desmoplastic variant of ameloblastomas in Malaysians. Br J Oral Maxillofac Surg. 1993; 31:299–303.

15. Takata T, Miyauchi M, Ogawa I, Zhao M, Kudo Y, Sato S, et al. So-called 'hybrid' lesion of desmoplastic and conventional ameloblastoma: report of a case and review of the literature. Pathol Int. 1999; 49:1014–1018.

16. Philipsen HP, Reichart PA, Takata T. Desmoplastic ameloblastoma (including "hybrid" lesion of ameloblastoma). Biological profile based on 100 cases from the literature and own files. Oral Oncol. 2001; 37:455–460.

17. Eversole LR, Leider AS, Strub D. Radiographic characteristics of cystogenic ameloblastoma. Oral Surg Oral Med Oral Pathol. 1984; 57:572–577.

18. Sun ZJ, Wu YR, Cheng N, Zwahlen RA, Zhao YF. Desmoplastic ameloblastoma - A review. Oral Oncol. 2009; 45:752–759.

19. Huang CM, Chen JY, Chen CH, Huang CJ. Radiotherapy for a repeatedly recurrent ameloblastoma with malignant transformation. Head Neck. (in press).

20. Thompson IO, van Rensburg LJ, Phillips VM. Desmoplastic ameloblastoma: correlative histopathology, radiology and CT-MR imaging. J Oral Pathol Med. 1996; 25:405–410.

21. Bianchi S, Tarello F, Polastri F, Valente G. Ameloblastoma of the mandible involving an autogenous bone graft. J Oral Maxillofac Surg. 1998; 56:1187–1191.

22. Stea G. Recurrence of an ameloblastoma in an autogenous iliac bone graft. J Oral Maxillofac Surg. 1985; 43:374–377.

23. 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.

24. Philipsen HP, Ormiston IW, Reichart PA. The desmo- and osteoplastic ameloblastoma. Histologic variant or clinicopathologic entity? Case reports. Int J Oral Maxillofac Surg. 1992; 21:352–357.

25. Effiom OA, Odukoya O. Desmoplastic ameloblastoma: analysis of 17 Nigerian cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2011; 111:e27–e31.

26. Kawai T, Kishino M, Hiranuma H, Sasai T, Ishida T. A unique case of desmoplastic ameloblastoma of the mandible: report of a case and brief review of the English language literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1999; 87:258–263.

27. Gardner DG, Heikinheimo K, Shear M, Philipsen HP, Coleman H. Ameloblastomas. In : Barnes L, Eveson JW, Reichart P, Sidransky D, editors. World Health Organization classification of tumors: pathology and genetics of head and neck tumors. 3rd ed. Lyon: IARC Press;2005. p. 296–300.

28. Keszler A, Paparella ML, Dominguez FV. Desmoplastic and non-desmoplastic ameloblastoma: a comparative clinicopathological analysis. Oral Dis. 1996; 2:228–231.

29. Ashman SG, Corio RL, Eisele DW, Murphy MT. Desmoplastic ameloblastoma. A case report and literature review. Oral Surg Oral Med Oral Pathol. 1993; 75:479–482.

30. Zachariades N. Recurrences of ameloblastoma in bone grafts. Report of 4 cases. Int J Oral Maxillofac Surg. 1988; 17:316–318.

31. Gold L, Upton GW, Marx RE. Standardized surgical terminology for the excision of lesions in bone: an argumentum for accuracy in reporting. J Oral Maxillofac Surg. 1991; 49:1214–1217.

32. Philipsen HP, Reichart PA. Unicystic ameloblastoma. A review of 193 cases from the literature. Oral Oncol. 1998; 34:317–325.