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J Stomatol Oral Maxillofac Surg. 2017 Dec;118(6):363-370. doi: 10.1016/j.jormas.2017.08.005. Epub 2017 Aug 30.

Clear cell odontogenic carcinoma. A review.

Author information

1
Department of Oral Surgery, Odontology, Timone Hospital, 13385 Marseille, France; Department of Stomatology, Oral & Maxillofacial surgery, Conception Hospital, 13005 Marseille, France; UMR 7268 ADES, Faculty of Medicine, Aix-Marseille University/EFS/CNRS, 13344 Marseille, France. Electronic address: mehdi.hadj-said@ap-hm.fr.
2
Department of Oral Surgery, Odontology, Timone Hospital, 13385 Marseille, France.
3
Department of Pathological Anatomy & Cytology, University Cancer Institute Toulouse Oncopole, 31059 Toulouse, France; Department of Plastic & Maxillofacial Surgery, Pierre-Paul Riquet Hospital, 31059 Toulouse, France.
4
Department of Pathological Anatomy & Cytology, University Cancer Institute Toulouse Oncopole, 31059 Toulouse, France.
5
Department of Stomatology, Oral & Maxillofacial surgery, Conception Hospital, 13005 Marseille, France.
6
Department of Oral Surgery, Odontology, Timone Hospital, 13385 Marseille, France; UMR 7268 ADES, Faculty of Medicine, Aix-Marseille University/EFS/CNRS, 13344 Marseille, France.

Abstract

INTRODUCTION:

Clear cell odontogenic carcinoma (CCOC) is described as an exceptional and hard to diagnose malignant tumor which was first reported by Hansen in 1985. The purpose of this review article is to show that CCOC is a not that rare entity and to discuss its various aspects in order to enhance the diagnosis.

MATERIAL AND METHODS:

A search in the English language literature was performed using the Scopus, ScienceDirect, PubMed and Medline databases between 1985 and 2016. Data were collected on epidemiologic, clinical, radiographic, histological, immunohistochemistrical, cytogenetic, management, follow-up and prognosis features of CCOC.

RESULTS:

Sixty-five studies from which a total of 95 case reports were included in the review. CCOC was generally seen in the fifth decade and the most common site was mandibular. The most frequently found symptoms were swelling, tooth mobility and pain. Radiologically, the image was radiolucent and could look like a cyst or a periodontal lesion. In situ hybridization techniques frequently expressed a gene fission of EWSR1. The treatment was mostly a radical surgical excision of the tumor with or without adjuvant radiotherapy or chemotherapy. CCOC showed high rates of recurrence and mortality related with the presence of distance metastasis.

DISCUSSION:

Fission of EWSR1 gene could be the main element it the diagnosis of CCOC. A multidisciplinary approach, including a radiologist, pathologist and an oral & maxillofacial surgeon may be helpful in the evaluation and management of these lesions. With 95 reports found in English literature, we cannot say that CCOC is extremely rare anymore.

KEYWORDS:

Clear cells; Immunohistochemistry; Odontogenic tumors; Oral cancers

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