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      Malignant transformation of calcifying cystic odontogenic tumour – a review of literature

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          Abstract

          Calcifying cystic odontogenic tumour (CCOT) has been classified as an odontogenic tumour. Ghost cell odontogenic carcinoma (GCOC) is the malignant counterpart of CCOT. This paper aims to review the literature regarding malignant transformation of CCOT.

          A literature search was done via the National Library of Medicine PubMed interface, searching for articles relating to malignant transformation of CCOT. From these articles, references were obtained, and from their references lists, pertinent secondary references were also identified and acquired.

          After reviewing the literature, we found 26 cases of GCOC which developed from CCOT. Malignant transformation of CCOT was seen more commonly in the maxilla. Histologically, changes such as increased nuclear/cytoplasmic ratio, atypical mitotic figures have been reported after malignant transformation. Immunohistochemical analysis has shown an increased expression of ki-67 and p53 in tumour cells.

          Malignant transformation of CCOT, although rare, mostly takes place in recurrent and long standing cases.

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          Most cited references24

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          International collaborative study on ghost cell odontogenic tumours: calcifying cystic odontogenic tumour, dentinogenic ghost cell tumour and ghost cell odontogenic carcinoma.

          Calcifying odontogenic cyst was described first by Gorlin et al. in 1962; since then several hundreds of cases had been reported. In 1981, Praetorius et al. proposed a widely used classification. Afterwards, several authors proposed different classifications and discussed its neoplastic potential. The 2005 WHO Classification of Odontogenic Tumours re-named this entity as calcifying cystic odontogenic tumour (CCOT) and defined the clinico-pathological features of the ghost cell odontogenic tumours, the CCOT, the dentinogenic ghost cell tumour (DGCT) and the ghost cell odontogenic carcinoma (GCOC). The aim of this paper was to review the clinical-pathological features of 122 CCOT, DGCT and GCOC cases retrieved from the files of the oral pathology laboratories from 14 institutions in Mexico, South Africa, Denmark, the USA, Brazil, Guatemala and Peru. It attempts to clarify and to group the clinico-pathological features of the analysed cases and to propose an objective, comprehensive and useful classification under the 2005 WHO classification guidelines. CCOT cases were divided into four sub-types: (i) simple cystic; (ii) odontoma associated; (iii) ameloblastomatous proliferating; and (iv) CCOT associated with benign odontogenic tumours other than odontomas. DGCT was separated into a central aggressive DGCT and a peripheral non-aggressive counterpart. For GCOC, three variants were identified. The first reported cases of a recurrent peripheral CCOT and a multiple synchronous, CCOT are included. Our results suggest that ghost cell odontogenic tumours comprise a heterogeneous group of neoplasms which need further studies to define more precisely their biological behaviour.
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            Ghost cell odontogenic carcinoma transformed from a dentinogenic ghost cell tumor of maxilla after multiple recurrences.

            Dentinogenic ghost cell tumor (DGCT), also referred to as odontogenic ghost cell tumor, is a rare tumor classified as a neoplastic variant of calcifying odontogenic cyst (COC). Ghost cell odontogenic carcinoma (GCOC) is the especially rare malignant counterpart of DGCT and COC. The case of a middle-aged male with a maxillary DGCT which transformed to GCOC after 5 recurrences during a 21-year period is presented. When the diagnosis of GCOC (first identified as malignant after 17-year recurrence) was made, the tumor cells showed more obvious atypia, necrosis, and numerous mitoses, and interestingly the ghost cell was hard to find and even disappeared. The significances in pathologic diagnosis and clinical treatment are discussed.
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              Clinicopathologic spectrum of the so-called calcifying odontogenic cysts: a study of 21 intraosseous cases with reconsideration of the terminology and classification.

              Tie Li, Shi Yu (2003)
              The so-called calcifying odontogenic cyst (COC) represents a heterogeneous group of lesions that exhibit a variety of clinicopathologic and behavioral features. Because of this diversity, there has been confusion and disagreement on the terminology and classification of these lesions. We reviewed the clinicopathologic features of 21 intraosseous cases that were previously diagnosed as COC or under related diagnostic terms. Based on the biologic behavior, the lesions of the present series were divided into three subgroups: cyst, benign tumor, and malignant tumor. Sixteen cases (nine men and seven women) proved to be unicystic lesions with (five cases) or without associated odontoma. The lining epithelium of the cystic lesions fulfilled the histologic criteria for COC proposed by the World Health Organization, and their overall clinicopathologic features were consistent with that of developmental odontogenic cysts. The age of patients from the cyst group peaked at the second decade. The maxilla was affected more often (69%) than the mandible, with a predilection for the canine-premolar region (62.5%). Thirteen patients with follow-up information revealed no recurrence following enucleation. The four cases in the benign tumor group had variable clinicopathologic features. Two cases were solid tumors consisting of ameloblastoma-like sheets of odontogenic epithelium that contained ghost cells/calcification foci and juxtaepithelial dentinoid. Both patients experienced multiple recurrences following conservative surgeries. The other two lesions contained typical areas of COC and other types of odontogenic tumors (one ameloblastoma and one odontogenic myxofibroma). All four lesions occurred in the mandible and were relatively large. In the present series one case identified as malignant tumor arose from a previously benign COC. The tumor shared some features of COC (ghost cell foci and dystrophic calcification) but also had prominent mitotic activity, nuclear and cytoplasmic pleomorphism, areas of tumor necrosis, and infiltrative/destructive growth. Recognizing the extreme diversity in clinicopathologic features and biologic behavior among the so-called COCs, we suggest that the term COC should be used to specifically designate the unicystic lesions with or without an associated odontoma, i.e., lesions of the cyst group, and other related lesions identified as benign tumor and malignant tumor should be termed and classified separately. A tentative scheme with respect to the terminology and classification for this group of disparately behaving lesions was herein proposed to reflect the likely difference of their nature.
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                Author and article information

                Journal
                Contemp Oncol (Pozn)
                Contemp Oncol (Pozn)
                WO
                Contemporary Oncology
                Termedia Publishing House
                1428-2526
                1897-4309
                08 July 2015
                2015
                : 19
                : 3
                : 184-186
                Affiliations
                [1 ]Department of Oral Maxillofacial Sciences, Al-Farabi College of Dentistry and Nursing, Riyadh, Saudi Arabia
                [2 ]Department of Restorative Dentistry, Al-Farabi College of Dentistry and Nursing, Riyadh, Saudi Arabia
                Author notes
                Address for correspondence: Bassel Tarakji, Department of Oral Maxillofacial Sciences, Al-Farabi College of Dentistry and Nursing, Riyadh, Saudi Arabia. e-mail: denpol@ 123456yahoo.co.uk
                Article
                25406
                10.5114/wo.2015.52654
                4631291
                a310f894-6d08-4635-b3fa-f656a50cd74d
                Copyright © 2015 Termedia

                This is an Open Access article distributed under the terms of the Creative Commons Attribution-Noncommercial 3.0 Unported License, permitting all non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 09 March 2014
                : 05 June 2014
                : 06 August 2014
                Categories
                Review

                calcifying cystic odontogenic tumour,malignancy,ghost cell odontogenic tumour,review,gorlin cyst,odontogenic cyst

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