BUJOD 2013-c final

BUJOD Case Report Ameloblastic Carcinoma associated with impacted tooth - A rare case report Authors : Gouse Mohiddin*, Shantha Bharathan**, B.Thayumanavan***, S.Rajkumari****, Prashant Kumar***** ABSTRACT: Oral cancer in general refers to squamous cell carcinoma, while odontogenic tumors and other tumors of the oropharyngeal region are of least significance because of its rare occurrence. However the odontogenic tumors pose a great difficulty in diagnosis since the carcinomas of odontogenic origin exhibit a varied clinical and histopathological picture. We encountered a male patient with a swelling associated with missing teeth which on wary scrutinization turned out to be an odontogenic carcinoma. Key words: ameloblastoma, carcinoma, impaction, malignant INTRODUCTION: Odontogenic carcinomas are the rarest entity among the malignant tumors, as very few cases have been reported in English literature. These carcinomas are thought to arise de novo or from preexisting benign odontogenic tumors exhibiting some resemblance to the primary tumor however dedifferentiation can be appreciated.(1,2) Malignancy associated with ameloblastoma can be categorized as ameloblastic carcinoma or malignant ameloblastoma where the former presents with cytological atypia with or without metastasis while the later presents a definite metastasis without (3,4) cytological atypia. Ameloblastic carcinomas seem to be more common compared to malignant (5) ameloblastoma in the ratio 2:1. CASE REPORT A 17 year old male presented with a painless swelling in left on the left side of the face corresponding to the angle of mandible, on intra oral examination the patient presented with swelling over the left mandibular posterior region with expansion of buccal and lingual cortical plates, Address for correspondence Dr. Gouse Mohiddin, M.D.S., Reader, Kalinga institute of dental sciences e-mail address: [email protected]

obliterating the buccal vestibule, missing of 37 was noted. On palpation the swelling was bony hard and tender. Orthopantomographic X-ray revealed radiolucency associated with impacted 37, 38 extending across the midline upto 42, with root resorption in relation to 36, 35, 34, 33, 32,31,41. 38 and 37 are impacted. The lower border of the mandible is thinned out. Concluding with these features a provisional diagnosis was drawn as Odontogenic keratocyst. The impacted teeth were removed and the cystic lesion was enucleated submitted for histopathological confirmation. Microscopic evaluation revealed anastomosing cords of odontogenic epithelium consisting of central loosely arranged cells resembling stellate reticulum type of cells and peripheral tall columnar cells. In between the odontogenic epithelial strands is the connective tissue stroma. Based on these findings the lesion was confirmed as unicystic ameloblastoma plexiform type . The patient was planned was mandiblectomy followed by reconstruction. Gross examination of the excised mandible revealed perforation on the buccal as well as the lingual cortical plates at the level of premolar. From the excised specimen serial sections were made and evaluated. Low power view (10X) revealed odontogenic epithelium

*Reader, Kalinga institute of dental sciences, Orissa **, ***Professor, **** Sr. Lecturer Sathyabama university dental college and hospital, Chennai *****Senior Lecturer, HKEs S.N.Institute of Dental Sciences & Research, Gulbarga

BHAVNAGAR UNIVERSITY'S JOURNAL OF DENTISTRY

Vol. 3 Issue-1 Jan. 2013

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BUJOD arranged in interconnecting strands with peripheral layers resembling squamous type of cells and central loosely arranged stellate type of cells. On closer examination (40X) the peripheral layer of cells revealed tall columnar cells admixed with squamous type of cells exhibiting cellular atypia such as cellular pleomorphism and nuclear hyperchromatism while the central cells were stellate type mixed exhibiting squamous metaplasia. Immunohistochemical evaluation of the sections that revealed squamous metaplasia and dysplastic features with Ki67 – a proliferative marker disclosed moderate expression. These findings warrant a diagnosis of ameloblastic carcinoma. DISCUSSION: Ameloblastoma associated with impacted tooth is (6 ) mostly unicystic variety. Unicystic ameloblastoma .(7) may be luminal or mural type The epithelium lining the cyst does not always satisfy the Vickers and Gorlin's criteria however it may present plexiform pattern of odontogenic epithelium which can be described as plexiform unicystic (8) ameloblastoma. In our case study, the radiographic appearance was consistent with a cystic lesion which on further microscopic investigation confirmed unicystic ameloblastoma exhibiting plexiform pattern, however perforation of the cortical plates is characteristic feature of malignancy that lead to critical evaluation of the excised specimen by preparing serial sections which turned out as a ameloblastic carcinoma. The carcinoma arising within the bone must be assessed in terms of primary intra-alveolar carcinoma if there is any feature of ameloblastomatous area. If there is ameloblastoma associated with the carcinoma then keratoameloblastoma and acanthomatous variant of ameloblastoma must be ruled out.(9) However the variants of ameloblastoma do not present with cytological alterations.

Mohiddin et al

that ameloblastoma can present a wide range of histological picture. Hence it is mandatory that all the parts of biopsied tissue must be evaluated for better understanding of the disease that may provide a better prognosis. REFERENCES: 1.

Sylvie Louise Avon, John McComb, Cameron Clokie, Ameloblastic carcinoma: Case report and literature review, Journal of the Canadian Dental Association, 2003;69(9):573-576

2.

Hye-Jung Yoon, Sam-Pyo Hong, Jae-Il Lee, Sam-Sun Lee, Seong-Doo Hong, Seoul, Ameloblastic carcinoma: an analysis of 6 cases with review of literature, Oral Surg Oral Med Oral Pathol, 2009;108(6):904-913

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Maya Ramesh B. Sekar S. Murali Saramma Mathew James Chacko George Paul, Ameloblastic Carcinoma –review and histopathology of 5 cases, Oral and Maxillofacial Pathology Journal, 2011; 2 (2):154-160.

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Reichart. P.A, Philipsen .H.P- Odontogenic Tumours and Allied Lesions, 2004 Quintessence Radiology and Endodontology 2006; 101: 5 :638- Publishing Co Ltd,London, ISBN 1-85097059-9

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Rajendran R, Sivapathasundharam B, Shafer's Textbook Of Oral Pathology, Elsevier, India, 2006, fifth edition

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Renuka Ammanagi, Vaishali Keluskar, Rakhi Issrani, Plexiform Unicystic Ameloblastoma Report Of A Case With Unusual Presentation, Int J Dent Case Reports 2011; 1(1): 1-10

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Gardner DG, Washington DC, Corio RL. The relationship of plexiform unicystic ameloblastoma to conventional ameloblastoma. J Oral Surg 1983;56:54-60.

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Corio LR, Goldblatt LI, Edwards PA, Hartman KS. Ameloblastic carcinoma: a

CONCLUSION: This case is so unique that it posed as a innocent cystic lesion at the initial stage of diagnosis which later turned out to be a carcinoma, which suggests



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BUJOD clinicopathologic study and assessment of eight cases. Oral Surg Oral Med Oral Pathol 1987; 64(5):570–6.

Figure 4(b) High power view of metaplastic area revealing dysplastic features

Figure 2 Photomicrograph (10X) shows plexiform pattern of ameloblastoma with cystic degeneration in the connective tissue stroma

Figure 5 (a) Low power photomicrograph reveals Ki67 positivity in dysplastic areas

Figure 3 Exicised specimen showing perforation of the buccal cortical plate

Figure 5(b) High power view of Ki67 positive areas

Figure 4(a) Low power photomicrograph reveals squamous metaplasia


Source of Support :

NIL

Conflict of Interest :

NOT DECLARED

Date of Submission :

10-09-2012

Review Completed :

11-12-2012


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