IRUCAA@TDC : Adenomatoid odontogenic tumor arising from the mandibular molar region: a case report and review of the literature
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(2) 223. Bull. Tokyo dent. Coll., Vol. 45, No. 4, pp. 223⬃227, November 2004. Case Report. ADENOMATOID ODONTOGENIC TUMOR ARISING FROM THE MANDIBULAR MOLAR REGION: A CASE REPORT AND REVIEW OF THE LITERATURE DAISUKE SATO, KENICHI MATSUZAKA, MITSURU YAMA*, TAKASHI KAKIZAWA* and TAKASHI INOUE Department of Clinical Pathophysiology, Tokyo Dental College, 1-2-2 Masago, Mihama-ku, Chiba 261-8502, Japan * Department of Oral and Maxillofacial Surgery, Suidobashi Hospital, Tokyo Dental College, 2-9-18 Misakicho, Chiyoda-ku, Tokyo 101-0061, Japan. Received 12 November, 2004/Accepted for Publication 1 February, 2005. Abstract This case report describes a rare case of an adenomatoid odontogenic tumor (AOT) arising in the mandibular third molar region of a 20-year-old female. The tumor was a well-circumscribed intraosseous lesion with an embedded tooth. Histologically, the induction of extensive globular calcification was evident in addition to the characteristic histological features of AOT. The present case lends support to the categorization of AOT as an odontogenic tumor consisting of a disorderly mixture of odontogenic epithelium and odontogenic ectomesenchyme with calcification. Key words:. Adenomatoid odontogenic tumor— AOT— Odontogenic tumor— Mandibular. INTRODUCTION Adenomatoid odontogenic tumors (AOT) are rare, slow growing, benign, odontogenic, epithelial tumors which usually arise in the second or third decade15,20). AOT are usually located in the anterior region of the maxilla; they normally, produce slow swelling without pain15,20). The tumor growth may cause displacement of teeth rather than root resorption4). AOTs comprise only 0.1% of tumors and cysts of the jaw5). Philipsen et al. reported that they account for 3 to 7% of all odonto-. genic tumors15). In Philipsen’s report, studies in Nigeria (Sawyer17)/Ajagbe et al.1)) showed a higher frequency, but other studies (Regezi et al.16) in the USA and Happonen et al.6) in Finland, among others) record a lower frequency. We herein report a rare case of AOT and review the literature.. CASE REPORT A 20-year-old female with a painless swelling of the mandibular third molar area was. 223.
(3) 224. D. SATO et al.. Fig. 1 Preoperative panoramic radiography showing a well circumscribed unilocular radiolucency around the third molar (arrowhead). admitted to the Tokyo Dental College Department of Oral Surgery. Clinical examination revealed a circumscribed, bucco-lingual swelling. Radiographic examination showed a unilocular radiolucency extending with an embedded tooth (Fig. 1). No root resorption or displacement of neighboring teeth was seen. The lesion was completely enucleated under the clinical diagnosis of a dentigerous cyst, and sent to the Department of Clinical Pathophysiology of the Tokyo Dental College for pathological diagnosis.. RADIOLOGIC FINDINGS A radiolucent lesion with a regular border was seen in the mandibular third molar area. The radiolucency associated with the unilocular legion extended to the apical regions of the unerupted teeth. No displacement of neighboring teeth due to tumor expansion or root resorption was seen (Fig. 1).. HISTOPATHOLOGICAL FINDINGS The lesion was a well-defined mass 22⳯18 mm in size that was surrounded by a thick, fibrous capsule. The mandibular third molar was encapsulated. After the specimen. Fig. 2 a. Dysplastic calcification H-E staining b. Duct-like and adenoid structure Arrowhead: Tall columnar cell. was fixed with 10% formalin, paraffin sections were prepared for light microscopy and stained with hematoxylin and eosin using routine methods. Histological examination revealed amorphous eosinophilic droplets and foci of dystrophic calcification scattered in and around the epithelial elements (Fig. 2). The epithelial elements consisted of polyhedral cells in loose and irregular arrangements, spindle cells, and tall columnar cells that formed duct-like structures as a result of the degeneration of the stromal tissue. The nuclei of the columnar cells were polarized away from the central space. The interstitium consisted of loosely arranged fibrous connective tissue containing dilated vessels, and a lot of small cystic spaces were present..
(4) ADENOMATOID ODONTOGENIC TUMOR IN THE MANDIBLE. 225. Table 1 9 cases of AOT was diagnosed from 1967 to 2004 at Tokyo Dental College Hospital Year. Age. Gender. Region. ’73. 20. F. Bone RtU 1–6. ’75. 40. F. Bone RtL34. ’75. 9. F. Bone RtU12. ’77. 17. M. Bone RtU3. ’79. 48. M. Bone LtL45. ’83. 14. F. Bone RtL23. ’84. 23. F. Bone LtL78. ’93. 20. F. Bone LtL32. ’98. 20. F. Bone RtL8 Present case Tokyo Dental College Hospital. DISCUSSION AOT is defined as a tumor of the odontogenic epithelium with duct-like structures and varying inductive changes in the connective tissue3–5,19). Preoperatively, AOTs have been diagnosed as various types of disease, because of the variety of clinical and radiographic features of the lesions. Radiographically, they frequently look like a dentigerous cysts or follicular cysts. The radiolucency associated with an adenomatoid odontogenic tumor may extend more apically than that of a dentigerous cyst5). The tumors are usually 1.5 to 3 cm5), but larger lesions have been reported in the literature9,21). The lesions usually appear unilocular radiographically, but at least five cases of multilocular appearance have been reported5,9). Larsson reported a case of multiple AOT-like jawbone lesions in a young patient 9). AOT lesions may often appear completely radiolucent; however, they contain fine calcifications7,21), a feature that may be helpful in differentiating AOT from dentigerous cysts. The calcified materials seen in AOT have been considered to be a form of enamel10), dentin22), enamel and dentin11), cementum18), dentin and cementum12) and dystrophic calcification7), but their exact nature remains con-. troversial19). In the present case, no dental hard tissue formation was observed; only dystrophic calcification was seen. Ishikawa and Mori suggested that most of the duct-like structures are formed by degeneration of the stromal tissue, because some of the luminal content had the same staining characteristics as the mesenchymal fibrous tissue7). A direct transition between the duct-like lumen and the degenerating stroma was clearly evident. The characteristic features of this tumor are probably due to both the highly differentiating ability of the epithelial component and the lack of mesenchymal tissue receptive to the influence of the epithelium. Information on the incidence and prevalence of individual odontogenic tumors is still not available. However, AOT is a particularly rare tumor that represents 3% to 7% of all odontogenic tumors2,5,8,14,15,21). In our laboratory, AOT was diagnosed in 9 cases during the period from 1967 to 2004 (Table 1), which comprises only 1.8% of odontogenic tumors (10/559) seen during that time. The age of patients with AOT ranges between 3 and 82 years of age at the time of diagnosis14,15), although those tumors are uncommon in patients older than 30. The age distribution peaks in the second.
(5) 226. D. SATO et al.. decade1,3,6,7,9,11); two-thirds of AOT cases are diagnosed in the second decade2,14,15). In our laboratory, only two cases have been older than 30. In Philipsen’s report, the ratio of patients at least 30 years old is 7.5% (25/ 332)15). Among Japanese cases, 10.5% of the patients are reported to be over 30 years old (13/126)20). The maxilla is involved nearly twice as frequently as the mandible15,20), and unerupted permanent teeth are associated with this lesion in one-third of the cases15,20). Three quarters of these lesions involve the anterior aspect of the jaw, and the canine area of the maxilla is the most common site of development5,14,20). In our laboratory, 3 cases of AOT were in the maxilla region and 6 cases in the mandible. It is remarkable that the mandible is more frequently involved than the maxilla and that 2 cases were found in the molar region of the mandible. All these cases were of the intraosseous type. AOT affects females more than males at almost a two to one ratio, according to the published English literature2–5,8,13,14). AOT-cases in Asians show a male:female ratio of 1: 2.3, whereas cases outside of Asia show a ratio of 1:1.4, a difference that is statistically significant15). In Japan, these lesions are found three times more frequently in females than in males3,14,15,20), which can not be explained at present15). In our laboratory, 2 cases were male and 7 cases were female (Table 1). Enucleation and currettage is the most common treatment modality for this tumor5,15,20,21), and recurrence is extremely rare. Only three cases of recurrent AOT among 750 cases have been reported, and all the recurrent cases were in the Japanese literature14,20). Careful follow-up examinations should be conducted in this disease.. REFERENCES 1) Ajagbe, H.A., Daramola, J.D., Junaid, T.A. and Ajagbe A.O. (1985). Adenomatoid odontogenic tumor in a black African population:. 2). 3) 4). 5). 6). 7). 8). 9). 10). 11). 12). 13). 14) 15). Report of thirteen cases. J Oral Maxillofacial Surg 43, 683–687. Brand, W.N., Douglas, D.D., Carl, M.A. and Jelly, E.B. (2002). Oral & Maxillofacial Pathology, 2nd ed., pp. 621–623, W.B. Saunders Company, Philadelphia. Cawson, R.A. and Odell, E.W. (2002). Oral Pathology and Oral Medicine, pp. 124–125, Churchill Livingstone, London. Engin, B., Fenda, T., Murat, A. and Sevket, R. (2001). An adenomatoid odontogenic tumor with unusual clinical features. J Oral Sci 43, 283–286. Ertunc, D., Gelengul, G., Murat, B. and Akif, C. (1997). Adenomatoid odontogenic tumor. Case report and review of the literature. Aust Dent J 42, 315–318. Happonen, R.P., Ylippaavalmiemi, P. and Calonius, B. (1982). A survey of 15,758 oral biopsies in Finland. Proc Finn Dent Soc 78, 201– 206. Ishikawa, G. and Mori, K. (1962). A histopathological study on the adenomatoid ameloblastoma. Report of four cases. Acta Odontol Scand 20, 419–432. Kramer, I.R., Pindborg, J.J. and Shear, M. (1992). Histological typing of odontogenic tumors, 2nd ed., In WHO International Histological Classification of Tumors. Springer-Verlag, Berlin, Heidelberg, New York. Larsson, A.K. and Swartz, K.H. (2003). A case of multiple AOT-like jawbone lesions in a young patient — a new odontogenic entity?. J Oral Pathol Med 32, 55–62. Mori, M., Makino, M. and Imai, K. (1989). The histochemical nature of homogenous amorphous materials in odontogenic epithelial tumors. J Oral Surg 38, 96–102. Moro, I., Okamura, N., Okuda, S., Komiyama, K. and Umemura, S. (1982). The eosinophilic and amyloid-like materials in adenomatoid odontogenic tumor. J Oral Pathol 11, 138–150. Nomura, M., Tanimoto, K., Takana, T. and Shimosato, T. (1992). Mandibular adenomatoid odontogenic tumor with unusual clinicopathologic features. J Oral Maxillofac Surg 50, 282–285. Philipsen, H.P., Srisuwan, T., Reichart, P.A. and Alcantara, S.P. (2002). Adenomatoid odontogenic tumor mimicking a periapical (radicular) cyst: A case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 94, 246–248. Philipsen, H.P. and Reichart, P.A. (1998). Adenomatoid odontogenic tumor: Facts and figures. Oral Oncol 35, 125–131. Philipsen, H.P., Reichart, P.A., Zhang, K.H., Nikai, H. and Yu, Q.X. (1991). Adenomatoid odontogenic tumor: Biologic profile based on.
(6) ADENOMATOID ODONTOGENIC TUMOR IN THE MANDIBLE. 499 cases. J Oral Pathol Med 20, 149–158. 16) Regezi, J.A., Kerr, D.A. and Courtney, R.M. (1978). Odontogenic tumors: Analysis of 706 cases. J Oral Surg 36, 771–778. 17) Sawyer, D.R. (1985). Oral pathology biopsy service in a developing country, Nigeria. Ann Dent 44, 42–49. 18) Spouge, J.D. (1967). The adenoameloblastoma. Oral Surg Oral Med Oral Pathol 23, 470–482. 19) Takeda, Y. and Shimono, M. (1996). Adenomatoid odontogenic tumor with extensive formation of tubular dentin. Bull Tokyo dent Coll 37, 189–193. 20) Toida, M., Hyodo, I., Okuda, T. and Tatematsu, N. (1990). Adenomatoid odontogenic tumor: Report of two cases in Japan. J Oral Maxillofac Surg 48, 404–408. 21) Vakur, O., Banu, G.K. and Cetin, K. (2003).. 227. Adenomatoid odontogenic tumor: A report of an unusual maxillary lesion. Quintessence International 34, 686–688. 22) Yamamoto, H., Kozama, Y., Hirai, G., Hagiwara, T. and Nakayama, T. (1981). Adenomatoid odontogenic tumor: Light and electron microscopic study. Int J Oral Surg 10, 272–278. Reprint requests to: Dr. Kenichi Matsuzaka Department of Clinical Pathophysiology, Tokyo Dental College, 1-2-2 Masago, Mihama-ku, Chiba 261-8502, Japan Tel: +81-43-270-3581 Fax: +81-43-270-3583 E-mail: [email protected].
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