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Case Report

Primary intraosseous carcinoma of the mandible arising from epithelial remnants of odontogenic cyst F. Spadari1, M. Rossi1, P. Salvatori2, R. Pertile1, G.P. Bombeccari1 of Biomedical, Surgical and Dental Sciences, Unit of Oral Pathology and Medicine, Ospedale Maggiore Policlinico Fondazione Ca’ Granda IRCCS, University of Milan, Italy 2Unit of Otolaryngology, Medical Management UO ORL AO Legnano, Legnano Hospital, Milan, Italy

1Department

Primary  intraosseous  squamous cell  carcinoma  (PIOSCC) is a rare malignant odontogenic tumor arising from odontogenic epithelial remnants within the jawbones. According to the 2005 WHO Classification of Tumors1, there are three subcategories of PIOSCC, histopathologically divided into 3 types: solid type carcinoma, carcinoma derived from a keratocystic odontogenic tumor, and carcinoma derived from an odontogenic cyst. We report a case of PIOSCC of the jaw with probable origin from rests of an odontogenic cyst. A 54-year-old woman complained pain and swelling on the left side of her neck and cheek mucosa three months later a two stages surgery for extraction of mandibular third molar associated with odontogenic cyst, in region of tooth #38. Panoramic radiography (PR) and Nuclear Magnetic Resonance Imaging (NMRI) results suggested an osteolytic mandibular tumor. An incisional biopsy was then performed, which indicated a moderately-differentiated (G2) squamous cell carcinoma, with invasion of sub-mucous tissues. Based on the clinical and pathological findings, a diagnosis of PIOSCC was made. The patient underwent to hemimandibulectomy, left neck dissection, and received chemotherapy and head-neck radiation. Tumor staging was classified as T4,N2,M0. Gardner2 has argued that the diagnosis of such a tumor arising from a cyst requires the presence of a transitional layer between the normal cyst epithelium and the carcinoma cells. The longstanding chronic inflammation might be the main predisposing factor for malignant transformation in the cyst epithelium.3 In this case, no transitional layer was visible, as a result of severe inflammation lasting over 3 months. We concluded that the tumor had probably arisen from the cyst because the two tissue types were so closely aligned in a single specimen characterized by chronic infiltration of lymphocytes, plasma cells, and crystals of cholesterol. References 1.

2. 3.

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Eversole LR, Siar CH, van der Waal I. Primary intraosseous squamous cell carcinomas. In: Barnes L, Evson JW, Reichart P, Sidransky D (eds) World Health Organization classification of tumors. Pathology and genetics of head and neck tumors. World Health Organization International Agency for Research on Cancer. IACR Press, Lyon, 2005; pp 290-291. Gardner AF. A survey of odontogenic cysts and their relationship to squamous cell carcinoma. Dent J 1975; 41:161-167. Schwimmer AM, Aydin F, Morrison N. Squamous cell carcinoma arising in residual odontogenic cyst: report of a case and review of literature. Oral Surg Oral Med Oral Pathol 1991; 72:218-21.

Annali di Stomatologia 2013; Suppl. 2: 1-48

Primary intraosseous carcinoma of the mandible arising from epithelial remnants of odontogenic cyst.

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