{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,27]],"date-time":"2026-09-27T12:00:10Z","timestamp":1790510410901,"version":"4.1.0"},"reference-count":15,"publisher":"MDPI AG","issue":"4","license":[{"start":{"date-parts":[[2022,4,18]],"date-time":"2022-04-18T00:00:00Z","timestamp":1650240000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Diagnostics"],"abstract":"<jats:p>Clear cell odontogenic carcinoma (CCOC) is a rare, aggressive epithelial neoplasm of the jaw first described in 1985 by Hansen and classified as an odontogenic malignant tumor in the 2005 WHO classification. To date, only 117 cases have been reported in the literature written in English. In this paper, we report the atypical presentation of a pericoronal localized tumor in the right mandibular wisdom tooth discovered during a routine radiologic examination. The patient, a 44-year-old healthy female, was referred by her general dental practitioner for examination of temporomandibular dysfunction and recurrent myofascial pain. Anamnesis and clinical examination did not suggest tumoral disease. The osteolytic lesion was removed, and histological examination revealed a clear cell odontogenic tumor (CCOT) of the right posterior lower jaw. Segmental lower jaw resection was performed, and a free iliac crest bone transplant was fixed with mandibular reconstruction plate. No cervical neck dissection was needed. A five-year follow-up examination shows excellent local and systemic recovery and no sign of tumor recurrence. The patient still suffers from bruxism and myofascial related pain, which are treated conservatively with a Michigan splint and physiotherapy. Based on this additional case, we review the literature and discuss the challenging diagnostic aspects, the unusual clinical presentation, and the treatment of CCOC.<\/jats:p>","DOI":"10.3390\/diagnostics12041019","type":"journal-article","created":{"date-parts":[[2022,4,18]],"date-time":"2022-04-18T22:04:02Z","timestamp":1650319442000},"page":"1019","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["Unusual Presentation of Clear Cell Odontogenic Carcinoma: Case Report and Literature Review"],"prefix":"10.3390","volume":"12","author":[{"given":"Ra\u00fal","family":"Verdeja","sequence":"first","affiliation":[{"name":"Department of Surgery, Service of Maxillofacial and Oral Surgery, University Hospital and Faculty of Medicine, 1205 Geneva, Switzerland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Edouard","family":"Stauffer","sequence":"additional","affiliation":[{"name":"Private Pathology Institute (Promed), 1700 Fribourg, Switzerland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9347-3736","authenticated-orcid":false,"given":"Tommaso","family":"Lombardi","sequence":"additional","affiliation":[{"name":"Unit Oral Medicine and Maxillo-Facial Pathology, Department of Surgery, Service of Maxillofacial and Oral Surgery, University Hospital and Faculty of Medicine, 1205 Geneva, Switzerland"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2022,4,18]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"115","DOI":"10.1002\/hed.2890080208","article-title":"Clear Cell Odontogenic Tumor\u2014A New Histologic Variant with Aggressive Potential","volume":"8","author":"Hansen","year":"1985","journal-title":"Head Neck Surg."},{"key":"ref_2","unstructured":"Barnes, L., Eveson, J.W., Reichart, P., and Sidransky, D. (2005). World Health Organisation Classification of Tumours. Pathology and Genetics of Head and Neck Tumours, IARC Press."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"1405","DOI":"10.1016\/j.ijom.2019.05.006","article-title":"Clear cell odontogenic carcinoma: A rare jaw tumor. A summary of 107 reported cases","volume":"48","author":"Guastaldi","year":"2019","journal-title":"Int. J. Oral. Maxillofac. Surg."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"284","DOI":"10.1186\/s12957-015-0693-4","article-title":"Mandibular Clear Cell Odontogenic Carcinoma","volume":"13","author":"Kwon","year":"2015","journal-title":"World J. Surg. Oncol."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"559","DOI":"10.4103\/0976-237X.169849","article-title":"Clinical Enigma: A Rare Case of Clear Cell Odontogenic Carcinoma","volume":"6","author":"Walia","year":"2015","journal-title":"Contemp. Clin. Dent."},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"743","DOI":"10.1002\/dc.23297","article-title":"Cytomorphology and Immunohistochemistry of a Recurrent Clear Cell Odontogenic Carcinoma with Molecular Analysis: A Case Report with Review of Literature","volume":"43","author":"Harbhajanka","year":"2015","journal-title":"Diagn. Cytopathol."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"1559","DOI":"10.1007\/s12253-019-00741-x","article-title":"Clear Cell Odontogenic Carcinoma: A Clinicopathological and Immunocytochemical Analysis","volume":"26","author":"Liu","year":"2020","journal-title":"Pathol. Oncol. Res."},{"key":"ref_8","doi-asserted-by":"crossref","unstructured":"Desai, A., Rivera, C.M., Faquin, W.C., Iafrate, A.J., Rivera, M.N., Jaquinet, A., and Troulis, M.J. (2021). Clear cell carcinoma: A comprehensive literature review of 254 cases. Int. J. Oral. Maxillofac. Surg., in press.","DOI":"10.1016\/j.ijom.2021.03.018"},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"2988","DOI":"10.1002\/1097-0142(19921215)70:12<2988::AID-CNCR2820701242>3.0.CO;2-V","article-title":"The WHO Histological Typing of Odontogenic Tumours. A Commentary on the Second Edition","volume":"70","author":"Kramer","year":"1992","journal-title":"Cancer"},{"key":"ref_10","unstructured":"Barnes, L., Eveson, J.W., Reichart, P., Sidransky, D., Bang, G., and Koppang, H. (2005). Clear Cell Odontogenic Carcinoma. World Health Organisation Classification of Tumors. Pathology & Genetics: Head and Neck Tumors, IARC Press."},{"key":"ref_11","doi-asserted-by":"crossref","unstructured":"Labrador, A.J.P., Marin, N.R.G., Valdez, L.H.M., Valentina, M.P., Sanchez, K.B.T., Ibazetta, K.A.R., Johan, B., and Cesar, A.V. (2021). Wright Clear Cell Odontogenic Carcinoma a Systematic Review. 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Korean Assoc. Oral Maxillofac. Surg."},{"key":"ref_15","unstructured":"E-Naggar, A.D., Chan, J.K.C., Grandis, J.R., Takata, T., and Slootweg, T.J. (2017). Clear Cell Odontogenic Carcinoma. World Health Organisation Classification of Head and Neck Tumours, IARC Press. [4th ed.]."}],"container-title":["Diagnostics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2075-4418\/12\/4\/1019\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,10]],"date-time":"2025-10-10T22:56:15Z","timestamp":1760136975000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2075-4418\/12\/4\/1019"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,4,18]]},"references-count":15,"journal-issue":{"issue":"4","published-online":{"date-parts":[[2022,4]]}},"alternative-id":["diagnostics12041019"],"URL":"https:\/\/doi.org\/10.3390\/diagnostics12041019","relation":{},"ISSN":["2075-4418"],"issn-type":[{"value":"2075-4418","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,4,18]]}}}