Sclerosing odontogenic carcinoma — review of all published cases: is it a justifiable addition as a malignancy? / Carcinoma odontogênico esclerosante - revisão de todos os casos publicados: sua classificação como tumor maligno é justificável?

Abstract Introduction Sclerosing odontogenic carcinoma was a new addition to the list of head and neck tumors by World Health Organization in 2017. This lesion has scarcely been reported and a lack of pathognomonic markers for diagnosis exists. Objective The aim of the study was to summarize findings from the available literature to provide up-to-date information on sclerosing odontogenic carcinoma and to analyse clinical, radiological, and histopathological features to obtain information for and against as an odontogenic malignancy. Methods We conducted a comprehensive review of literature by searching Pubmed, EBSCO and Web of Science databases, according to PRISMA guidelines. All the cases reported as sclerosing odontogenic carcinoma in English were included. Data retrieved from the articles were gender, age, clinical features, site, relevant medical history, radiographical findings, histopathological findings, immunohistochemical findings, treatments provided and prognosis. Results Mean age at diagnosis of sclerosing odontogenic carcinoma was 54.4 years with a very slight female predilection. Sclerosing odontogenic carcinoma was commonly reported in the mandible as an expansile swelling which can be asymptomatic or associated with pain or paraesthesia. They appeared radiolucent with cortical resorption in radiograph evaluation. Histologically, sclerosing odontogenic carcinoma was composed of epithelioid cells in dense, fibrous, or sclerotic stroma with equivocal perineural invasion. Mild cellular atypia and inconspicuous mitotic activity were observed. There is no specific immunohistochemical marker for sclerosing odontogenic carcinoma. AE1/AE3, CK 5/6, CK 14, CK19, p63 and E-cadherin were the widely expressed markers for sclerosing odontogenic carcinoma. Surgical resection was the main treatment provided with no recurrence in most cases. No cases of metastasis were reported. Conclusion From the literature available, sclerosing odontogenic carcinoma is justifiable as a malignant tumor with no or unknown metastatic potential which can be adequately treated with surgical resection. However, there is insufficient evidence for histological grading or degree of malignancy of this tumor.

Resumo Introdução O carcinoma odontogênico esclerosante é a nova adição à lista de tumores de cabeça e pescoço da Organização Mundial da Saúde em 2017. Essa lesão é pouco relatada e não há marcadores patognomônicos para o diagnóstico. Objetivo Resumir os achados da literatura disponível para fornecer informações atualizadas sobre o carcinoma odontogênico esclerosante e analisar as características clínicas, radiológicas e histopatológicas a favor e contra sua classificação como uma lesão odontogênica maligna. Método Uma revisão abrangente da literatura foi feita nos bancos de dados Pubmed, Ebsco e Web of Science, de acordo com as diretrizes do Prisma. Todos os casos relatados em inglês como carcinoma odontogênico esclerosante foram incluídos. Os dados recuperados dos artigos foram sexo, idade, características clínicas, sítio do tumor, histórico médico relevante, achados radiográficos, achados histopatológicos, achados imuno-histoquímico, tratamentos instituídos e prognóstico. Resultados A média de idade ao diagnóstico de carcinoma odontogênico esclerosante foi de 54,4 anos, com uma predileção muito leve pelo sexo feminino. Tumores do tipo carcinoma odontogênico esclerosante foram comumente relatados na mandíbula como um edema expansivo, que pode ser assintomático ou associado a dor ou parestesia. Eles têm aparência radiolucente com reabsorção cortical na radiografia. Histologicamente, o carcinoma odontogênico esclerosante é composto por células epitelioides em estroma denso, fibroso ou esclerótico com invasão perineural ambígua. Atipia celular leve e atividade mitótica imperceptível foram observadas. Não há um marcador imuno-histoquímico específico para SOC. AE1/AE3, CK 5/6, CK 14, CK19, p63 e E-caderina foram os marcadores amplamente expressos para carcinoma odontogênico esclerosante. A ressecção foi o principal tratamento feito sem recorrência na maioria dos casos. Nenhum caso de metástase foi relatado. Conclusão De acordo com a literatura disponível, é justificável classificar o carcinoma odontogênico esclerosante como um tumor maligno com nenhum ou desconhecido potencial metastático, que pode ser tratado adequadamente com ressecção cirúrgica. Entretanto, não há evidências suficientes para a graduação histológica ou de malignidade desse tumor.

Misdiagnosis of ameloblastoma in a patient with clear cell odontogenic carcinoma: a case report

Clear cell odontogenic carcinoma (CCOC), a rare tumor in the head and neck region, displays comparable properties with other tumors clinically and pathologically. In consequence, an incorrect diagnosis may be established. A 51-year-old male patient who was admitted to the Department of Oral and Maxillofacial Surgery at Pusan National University Dental Hospital was initially diagnosed with ameloblastoma via incisional biopsy. However, the excised mass of the patient was observed to manifest histopathological characteristics of ameloblastic carcinoma. The lesion was ultimately diagnosed as clear cell odontogenic carcinoma by the Department of Oral Pathology of Pusan National Dental University. Therefore, segmental mandibulectomy and bilateral neck dissection were performed, followed by reconstruction with fibula free flap and reconstruction plate. Concomitant chemotherapy radiotherapy was not necessary. The patient has been followed up, and no recurrence has occurred 6 months after surgery.

An unusual presentation of ghost cell odontogenic carcinoma: A case report with review of literature

Ghost cell odontogenic carcinoma (GCOC) is a malignant odontogenic epithelial tumor which is an exceedingly rare, highly aggressive, rapidly growing, and infiltrative tumor forming the malignant counterpart of long-standing benign cystic lesions coming in the spectrum of calcifying odontogenic cysts. To date, only a few cases have been reported in the medical literature. A case of unusual presentation of GCOC is presented and the clinical, histopathological, and immunohistochemical features are discussed along with a literature review. Our case report further emphasizes the bizarre biological behavior of this tumor and the need for strict long-term surveillance of the patients as metastasis to distant sites has been reported.

Clear cell odontogenic carcinoma of the mandible: a treatment strategy

Abstract Clear cell odontogenic carcinoma (CCOC) is a rare odontogenic tumor of the jaws, histologically characterized by the presence of agglomerates of cells with eosinophilic cytoplasm. The patient, a 62-year-old Caucasian woman, presented an intraosseous lesion in the mandibular symphysis. A clinical examination revealed a discrete volumetric increase with a hard consistency, palpable to extraoral and intraoral examinations. Imaging studies revealed an extensive radiolucent area, without defined limits, extending from the region of the right second premolar to the left canine. Incisional biopsy analysis indicated a diagnosis of CCOC. The treatment proposed was segmental resection of the mandible with a safety margin. After six months without recurrence, definitive mandibular reconstruction was performed using an iliac crest graft, followed by rehabilitation with implant-supported denture after five months. After three years of post-resection follow-up, the patient has shown no evidence of recurrence or metastasis. She continues to be under follow-up. To conclude, CCOC must be considered a malignant tumor with aggressive behavior. Previous studies have shown that resection with free margins is a treatment with a lower rate of recurrence. Nevertheless, long-term follow-up is necessary for such patients.

Ghost cell odontogenic carcinoma on right mandible and its respective surgical reconstruction: a case report

Calcifying cystic odontogenic tumor (CCOT) is defined as an odontogenic cyst-like benign neoplasm that characteristically contains several ghost cells, ameloblastoma-like epithelium, and occasional calcification. Ghost cell odontogenic carcinoma (GCOC), a malignant form of CCOT, is an exceptionally rare malignant tumor. In this report, we present a case of a 53-year-old man whose chief complaint was a solitary mass on the right mandible area. The mass was completely removed through an extraoral surgical approach and reconstructive surgery was performed in two phases.

Malignant Transformation of Radicular Cyst/Residual Cyst: A Review of Literature.

Aim: The aim of this study is to review the literature concerning malignant transformation of radicular cyst. Material and Method: A literature search using MEDLINE, accessed via the National Library of Medicine PubMed interface (http://www.ncbi.nlm.nih.gov/pubmed), searching for articles relating to the malignant transformation of radicular/residual cyst written in English from 1960-2013. Results: Our search identified only fourteen studies available to date in English written literature. No randomized controlled trials to assess the possibility of malignant transformation of radicular/residual cyst were in the literature. Conclusion: The development of malignancy from radicular /residual cysts is rare, however, it should always be considered as a differential diagnosis. The numbers of studied cases in literature are few but small number of case series & case reports are available. Also it is recommended that further work involving large series of tumors arising from radicular /residual cyst to determine the malignancy potential.

Dentinogenic Ghost Cell Tumor: A Case Report and Review of Literature

Ghost Cell Odontogenic Carcinoma Arising from Calcifying Cystic Odontogenic Tumor: A Case Report

Ghost cell odontogenic carcinoma (GCOC) is an exceptionally rare and malignant odontogenic tumor with aggressive growth characteristics. We describe a case of GCOC which was considerably derived from a previously resected calcifying cystic odontogenic tumor (CCOT). Cellular atypia, mitotic activity, Ki-67 labeling index and matrix metalloprotease-9 positive expression rate were all increased in the currently resected specimen compared to the initial one. This is a rare case of malignant transformation of CCOT to GCOC with respect to its histopathological and immunohistochemical findings.

Carcinoma odontogénico de células claras / Odontogenic carcinoma of clear cells

El carcinoma odontogénico de células claras es un raro tumor odontogénico que se presenta principalmente en la parte anterior de la mandíbula, mayormente en mujeres entre la quinta y séptima década de vida. Se caracteriza histológicamente por redes de células con citoplasma claro mezcladas con células que contienen citoplasma eosinófilo. Es sumamente agresivo y puede presentar metástasis local o a distancia. Al emplearse el tratamiento por enucleación y curetaje, frecuentemente da lugar a recurrencia, por lo que se aconseja el tratamiento radical de resección mandibular hasta una zona libre de lesión. El carcinoma odontogénico de células claras debe ser considerado en el diagnóstico diferencial de tumores mandibulares que presenten células claras para el establecimiento del plan de tratamiento adecuado. Se debe incluir el seguimiento a largo plazo del paciente. Se presenta el caso de un paciente femenino de 69 años de edad, con una lesión presente en la región presinfisaria derecha, sin afectación de ganglios linfáticos regionales o metástasis aparente. Se efectuó osteotomía segmentaria mandibular y reconstrucción con placa de titanio(AU)

Clear cell odontogenic carcinoma is a rare malignant odontogenic neoplasm, with a predilection for the anterior segment of the jaws, and presenting basically in women between the fifth and seventh decades of life. CCOC is composed histopathologycally by strands or groups of cells showing clear cytoplasm associated with cells showing eosinophilic cytoplasm and some times with tall columnar cells that resambled ameloblasts. CCOC is considered a low-grade malignancy but in some cases its behavior is aggressive giving local and distant metastasis, so the recommended treatment is a radical resection and follows the patient very close. CCOC should be diferenciated microscopically from other local and metastatic lesions that are composed with clear cells also, in order to render a proper diagnosis and treatment. We are presenting a clinical case corresponding to a 69 years old female patient, with a 10 year history of a slow growing anterior mandibular lesion without lymph node or distant metastasis, with a biopsy diagnosis of clear cell odontogenic carcinoma, the patient was treated by partial resection of the anterior mandible and reconstructed with a titanium plate(AU)

Primary intraosseous carcinoma on mandible: A case report

Primary intraosseous carcinoma (PIOC) is a rare odontogenic carcinoma defined as a squamous cell carcinoma arising within a jaw having no initial connection with the oral mucosa, and probably developing from residues of the odontogenic epithelium. PIOC appears more common in male than female, especially at posterior portion of the mandible. Radiographic features of PIOC show irregular patterns of bone destruction with ill defined margins. It could be sometimes misdiagnosed as the cyst or benign tumor because it shows well defined margins. If it couldn't be done appropriate treatment initially, PIOC shows extremely aggressive involvement, extensive local destruction and spreads to the overlying soft tissue. Therefore accurate diagnosis in early state is necessary. The diagnosis criteria proposed for PIOC are : (1) absence of ulcer formation, except when caused by other factors, (2) histologic evidence of squamous cell carcinoma without a cystic component or other odontogenic tumor cell, and (3) absence of another primary tumor on chest radiograph obtained at the time of diagnosis and during a follow-up period of more than 6 month(Suei et al., 1994).