Journal List > J Korean Assoc Oral Maxillofac Surg > v.37(6) > 1032513

J Korean Assoc Oral Maxillofac Surg. 2011 Dec;37(6):520-523. Korean.
Published online December 27, 2011.  https://doi.org/10.5125/jkaoms.2011.37.6.520
Copyright © 2011 by The Korean Association of Oral and Maxillofacial Surgeons
Keratoameloblastoma of the maxilla: a case report
Ji-Hoon Won,1 Hye-Young Na,1 Hyun-sil Kim,2 Jin Kim,2 Woong Nam,1,2 In-Ho Cha,1,2 and Hyung Jun Kim1,2
1Department of Oral and Maxillofacial Surgery, College of Dentistry, Yonsei University, Seoul, Korea.
2Department of Oral Cancer Research Institute, College of Dentistry, Yonsei University, Seoul, Korea.

Corresponding author: Hyung Jun Kim. Department of Oral and Maxillofacial Surgery, College of Dentistry, Yonsei University, 134, Shinchon-dong, Seodaemun-gu, Seoul 120-752, Korea. TEL: +82-2-2228-3132, FAX: +82-2-364-0992, Email: kimoms@yuhs.ac
Received July 05, 2011; Revised August 25, 2011; Accepted October 12, 2011.

Abstract

A keratoameloblastoma is a histologically variant of the ameloblastoma group, which varies in size and contains keratin material in the fibrous connective tissue among cystic lesions. A keratoameloblastoma is a rare disease with only 13 cases reported in the literature since Pindborg's first report in 1970. A 41-year-old man visited, complaining of pus discharged from the right maxilla. He had been diagnosed with an odontogenic keratocyst and was treated with cyst enucleation in the past. The clinical and radiology examination found evidence of recurrence and finally diagnosed him with keratoameloblastoma after enucleation and biopsy. This report discusses the clinical, radiological and histological characteristics of keratoameloblastoma and its treatment. In addition, we report another case of keratoameloblastoma that had transformed from an odontogenic keratocyst.

Keywords: Keratoameloblastoma; Maxilla; Papilliferous keratoameloblastoma; Odontogenic keratocyst

Figures


Fig. 1
The panoramic radiograph showing radiolucent lesion occupying right maxilla.
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Fig. 2
Axial and coronal computed tomography (CT) view. A. The axial CT scan shows well-defined and low attenuated cystic lesion about 3.5 cm sized in the right maxilla posterior region. Also, we can find marked expansion of the maxilla with perforation of cortical plates. B. The coronal scan shows elevation and thickening of right sinus mucosa of floor. The lesion is involved with nasal cavity.
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Fig. 3
Postoperative panoramic radiograph taken 2 months after decompression of cystic lesion. The lesion become smaller than preoperative panoramic radiograph.
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Fig. 4
Postoperative computed tomography taken 3 months after decompression of cystic lesion. The surgical site exhibits bone formation without evidence of disease. Also, the lesion is separated from nasal cavity and mucosa.
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Fig. 5
A. Typical epithelial lining of an odontogenic keratocyst (1996. 3.). B, C, D. Follicular epithelial structure containing layers of parakeratin. The follicle is lined by ameloblast-like colummar cells exhibiting hyperchromatism, palisading, reversed polarity. Several solid keratinized epithelial follicles present in stroma (2009. 6) (H&E staining, A: ×40, B, C, D: ×200).
Click for larger image

References
1. Whitt JC, Dunlap CL, Sheets JL, Thompson ML. Keratoameloblastoma: a tumor sui generis or a chimera? Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007;104:368–376.
2. Pindborg JJ. In: Pathology of the dental hard tissues. 2nd ed. Philadelphia: Saunders; 1970.
3. Altini M, Lurie R, Shear M. A case report of keratoameloblastoma. Int J Oral Surg 1976;5:245–249.
4. Norval EJ, Thompson IO, van Wyk CW. An unusual variant of keratoameloblastoma. J Oral Pathol Med 1994;23:465–467.
5. Altini M, Slabbert HD, Johnston T. Papilliferous keratoameloblastoma. J Oral Pathol Med 1991;20:46–48.
6. Siar CH, Ng KH. 'Combined ameloblastoma and odontogenic keratocyst' or 'keratinising ameloblastoma'. Br J Oral Maxillofac Surg 1993;31:183–186.
7. Said-al-Naief NA, Lumerman H, Ramer M, Kopp W, Kringstein GJ, Persenchino F, et al. Keratoameloblastoma of the maxilla. A case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997;84:535–539.
8. Takeda Y, Satoh M, Nakamura S, Ohya T. Keratoameloblastoma with unique histological architecture: an undescribed variation of ameloblastoma. Virchows Arch 2001;439:593–596.
9. Collini P, Zucchini N, Vessecchia G, Guzzo M. Papilliferous keratoameloblastoma of mandible: a papillary ameloblastic carcinoma: report of a case with a 6-year follow-up and review of the literature. Int J Surg Pathol 2002;10:149–155.
10. Vered M, Buchner A, Dayan D, Shteif M, Laurian A. Solid variant of odontogenic keratocyst. J Oral Pathol Med 2004;33:125–128.
11. Ide F, Mishima K, Saito I. Solid-cystic tumor variant of odontogenic keratocyst: an aggressive but benign lesion simulating keratoameloblastoma. Virchows Arch 2003;442:501–503.
12. Odell EW, Morgan PR. In: Biopsy pathology of the oral tissues. London: Chapman & Hall Medical; 1998.
13. Partridge M, Towers JF, Towers JF. The primordial cyst (odontogenic keratocyst): its tumour-like characteristics and behaviour. Br J Oral Maxillofac Surg 1987;25:271–279.
14. Shear M. The aggressive nature of the odontogenic keratocyst: is it a benign cystic neoplasm? Part 1. Clinical and early experimental evidence of aggressive behaviour. Oral Oncol 2002;38:219–226.
15. Shear M. The aggressive nature of the odontogenic keratocyst: is it a benign cystic neoplasm? Part 2. Proliferation and genetic studies. Oral Oncol 2002;38:323–331.