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Rare clear cell odontogenic carcinoma associated with impacted tooth in a young patient: case report and literature review

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Abstract

Clear cell odontogenic carcinoma (CCOC) is a rare malignant odontogenic tumor. It is characterized by showing, on histopathological examination, clusters of vacuolated and clear tumor cells with epithelial differentiation surrounded by fibrocollagenous stroma and fibroblasts. The present study presents a rare clinical case of mandibular CCOC associated with an impacted tooth in a 26-year-old woman surgically treated with mandibulectomy and reconstruction with iliac crest bone graft. The patient has been followed up for 22 months without signs of recurrence. A search for case report/case series was carried out in the PUBMED database, as well as in the references of relevant previously published literature reviews. Ninety-six publications were identified, totaling 136 distinct cases reported. Female sex was the most affected (63.1%) with 63.3% of cases occurring in patients in the fifth, sixth, or seventh decades of life. The mandible was more affected than the maxilla (74.2%). Association of CCOC with impacted teeth was found in 2.4% of cases, thus rendering it a rare occurrence. The present case report corroborates the results of the survey regarding sex and anatomical location of the tumor; however, it contradicts the findings regarding age predilection. The case described is the fourth known occurrence of tooth impaction associated with the tumor and the first in a female. In conclusion, CCOC should be considered, as well as other malignancies, as a possible diagnosis of maxillary or mandibular intraosseous lesions even in unusual circumstances such as in association with impacted teeth and in young patients.

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Authors and Affiliations

  1. Departament of Dental Clinic, Federal University of Ceara, Fortaleza, Ceará, Brazil

    Erasmo Bernardo Marinho, Ana Paula Negreiros Nunes Alves, Mário Rogério Lima Mota & Fabricio Bitu Sousa

  2. Dental Specialties Center, CEO Municipal de Caucaia, Caucaia, Ceará, Brazil

    Erasmo Bernardo Marinho

  3. Head and Neck Surgery Service, Walter Cantídio University Hospital and Santa Casa de Misericórdia de Fortaleza, Fortaleza, Ceará, Brazil

    Francisco Januário Farias Pereira-Filho

  4. School of Dentistry, Christus University Center – UNICHRISTUS, Fortaleza, Ceará, Brazil

    Antonio Ernando Carlos Ferreira-Junior & Fabricio Bitu Sousa

Authors
  1. Erasmo Bernardo Marinho
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  2. Ana Paula Negreiros Nunes Alves
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  3. Francisco Januário Farias Pereira-Filho
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  4. Antonio Ernando Carlos Ferreira-Junior
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  5. Mário Rogério Lima Mota
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  6. Fabricio Bitu Sousa
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Contributions

All authors read, discussed, and approved the final manuscript. MARINHO, EB collected the literature data, prepared tables, obtained preoperative clinical photos and imaging exams, conducted initial clinical management, and obtained and wrote the manuscript. ALVES, APNN and MOTA, MRL discussed and made the histopathological diagnosis. PEREIRA-FILHO, FJF obtained postoperative clinical photos and imaging exams and discussed and performed the surgical treatment. FERREIRA-JUNIOR, AEC obtained and described histopathological and immunohistochemical images and prepared the figures. SOUSA, FB coordinated the study and reviewed the manuscript.

Corresponding author

Correspondence to Erasmo Bernardo Marinho.

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Ethics approval

This case report (registration number 3,511,864) was analyzed and approved by the Research Ethics Committee of the Federal University of Ceará, Brazil. Permission to use clinical images and complementary exams was obtained with the patient’s signature. The present case report was carried out following the Declaration of Helsinki (1964), as well as guidelines and regulatory standards of the Brazilian National Health Council/Ministry of Health (Resolution No. 466/2012).

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The authors declare that they have no conflict of interest.

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Marinho, E.B., Alves, A.P.N.N., Pereira-Filho, F.J.F. et al. Rare clear cell odontogenic carcinoma associated with impacted tooth in a young patient: case report and literature review. Oral Maxillofac Surg 26, 491–503 (2022). https://doi.org/10.1007/s10006-021-01006-1

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