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      Hybrid ameloblastoma and central giant cell lesion: Challenge of early diagnosis

      case-report

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          Abstract

          Hybrid lesions encompass the occurrence of different entities in one lesion. A 67-year-old woman was referred to the Oral and Maxillofacial Surgery Service for treatment of mandibular Central Giant Cell Lesion (CGCL) previously diagnosed. Intraoral examination revealed edentulism and a painless swelling extending from the alveolar ridge to the buccal vestibule with hard consistency covered by normal mucosae, with unknown duration. Panoramic radiograph revealed a large, multilocular and well-defined radiolucent lesion extending from the region of left mandibular lateral incisor teeth to right mandibular first molar with no evidence of osseous perforation. Initially, a treatment with intralesional injection of corticosteroids was performed. After 18 months of treatment, an increase in size of the osteolytic lesion was noted. An incisional biopsy was carried out and the microscopic examination revealed a unicystic ameloblastoma associated to CGCL. It was performed marsupialization and later the enucleation of residual lesion. The follow-up remains being performed.

          Key words:Hybrid lesion, central giant cell lesion, ameloblastoma.

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          Most cited references15

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          Central giant cell granuloma of the jaw: a review of the literature with emphasis on therapy options.

          Central giant cell granuloma (CGCG) is a benign lesion of the jaws with an unknown etiology. Clinically and radiologically, a differentiation between aggressive and non-aggressive lesions can be made. The incidence in the general population is very low and patients are generally younger than 30 years. Histologically identical lesions occur in patients with known genetic defects such as cherubism, Noonan syndrome, or neurofibromatosis type 1. Surgical curettage or, in aggressive lesions, resection, is the most common therapy. However, when using surgical curettage, undesirable damage to the jaw or teeth and tooth germs is often unavoidable and recurrences are frequent. Therefore, alternative therapies such as injection of corticosteroids in the lesion or subcutaneous administration of calcitonin or interferon alpha are described in several case reports with variable success. Unfortunately, randomized clinical trials are very rare or nonexistent. In the future, new and theoretically promising therapy options, such as imatinib and OPG/AMG 162, will be available for these patients.
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            Multinucleated giant cells in various forms of giant cell containing lesions of the jaws express features of osteoclasts.

            The nature and the mechanism involved in the formation of the multinucleated giant cells (MGCs) in various giant cell-containing lesions of the jaws are not fully understood. The aim of this study is to clarify the osteoclastic features of the MGCs in central giant cell granuloma (CGCG), peripheral giant cell granuloma (PGCG), cherubism, and aneurysmal bone cyst (ABC), and the mechanism underlying the interrelations between cellular components in the formation of the MGCs. Immunohistochemical study with a panel of antibodies including vacuolar H+-ATPase (V-ATPase), carbonic anhydrase II (CA II), Cathepsin K, matrix metalloproteinases-9 (MMP-9), CD68, and proliferating cell nuclear antigen (PCNA), and enzyme histochemical staining for tartarate-resistant acid phosphatase (TRAP) were applied on a total number of 53 cases of giant cell-containing lesions including CGCG (n = 34), PGCG (n = 6), cherubism (n = 7), and ABC (n = 6). In situ hybridization was also carried out to detect the mRNA expression of the receptor activator of NF-kappaB ligand (RANKL), a newly identified cytokine that is shown to be essential in the osteoclastogenesis, its receptor RANK (receptor activator of NF-kappaB ligand), and its decoy receptor OPG (osteoprotegerin) in these four types of lesions. Immunohistochemical and enzyme histochemical studies showed that both the MGCs and a fraction of mononuclear cells in these lesions were strongly positive for TRAP, V-ATPase, CA II, Cathepsin K, MMP-9, and CD68, while the spindle-shaped mononuclear cells were positive for PCNA. The results with in situ hybridization indicated that RANKL mRNA was mainly expressed in the spindle mononuclear cells while OPG was extensively distributed in both the MGCs and the mononuclear cells. RANK mRNA was expressed in the MGCs and some round mononuclear cells. These results suggest that MGCs in the four types of giant cell-containing lesions of the jaws show characteristics of the osteoclast phenotype. The mononuclear stromal cells, which show TRAP positively, may be the precursors of the MGCs. RANKL, OPG, and RANK expressed in these lesions may play important roles in the formation of the MGCs. The similar characteristics and mechanisms in the differentiation of MGCs in these lesions also suggest that there might be a similar kind of pathogenesis involved in the formation of the MGCs in these lesions
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              Intralesional injection of triamcinolone hexacetonide as an alternative treatment for central giant-cell granuloma in 21 cases.

              Central giant-cell granulomas are benign, but occasionally aggressive, lesions that traditionally have been treated surgically. 21 cases of central giant-cell granuloma of the jaw were treated with intralesional injection of corticosteroids. The treatment protocol adopted was intralesional injection of 20mg/ml triamcinolone hexacetonide diluted in an anaesthetic solution of 2% lidocaine/epinephrine 1:200,000 in the proportion 1:1; 1.0ml of the solution was infiltrated for every 1cm(3) of radiolucid area of the lesion, totalling 6 biweekly applications. Ten patients had aggressive lesions and 11 nonaggressive. Two patients showed a negative response to the treatment and underwent surgical resection, 4 showed a moderate response and 15 a good response. 8 of the 19 who had a moderate-to-good response to the drug treatment underwent osteoplasty to reestablish facial aesthetics. In these cases, only mature or dysplastic bone was observed, with the presence or absence of rare giant multinucleated cells. The advantages of this therapy are its less-invasive nature, the probable lower cost to the patient, lower risk and the ability to treat the lesion surgically in the future, if necessary. Copyright © 2010 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
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                Author and article information

                Journal
                J Clin Exp Dent
                J Clin Exp Dent
                Medicina Oral S.L.
                Journal of Clinical and Experimental Dentistry
                Medicina Oral S.L.
                1989-5488
                1 February 2020
                February 2020
                : 12
                : 2
                : e204-e208
                Affiliations
                [1 ]Postgraduate Program in Dentistry, Federal University of Santa Catarina, Florianopolis, Santa Catarina, Brazil
                [2 ]Bucomaxillofacial Residence Program, University Hospital, Federal University of Santa Catarina, Florianopolis, Santa Catarina, Brazil
                [3 ]Department of Pathology, Federal University of Santa Catarina, Florianopolis, Santa Catarina, Brazil
                Author notes
                Department of Pathology Federal University of Santa Catarina University Campus Trindade Florianópolis, Santa Catarina Brazil. Zip code: 88040-900 , E-mail: rogeriogondak@ 123456yahoo.com.br
                Article
                56441
                10.4317/jced.56441
                7018476
                d5fd9d29-1c65-4cbd-858d-673dd6f13666
                Copyright: © 2020 Medicina Oral S.L.

                This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 9 December 2019
                : 15 October 2019
                Categories
                Case Report
                Oral Medicine and Pathology

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