Case of Metastatic Clear Cell Odontogenic Carcinoma with Response to Chemoimmunotherapy

Overview

Journal BMJ Case Rep

Specialty General Medicine

Date 2024 Apr 29

PMID 38684356

Authors

Ian J Robertson

Quinn C Robbins

Peter C Li

Affiliations

Soon will be listed here.

Abstract

Our patient initially presented with 6 months of left jaw pain and gingival bleeding, leading to the discovery of a radiolucent left maxillary mass on dental evaluation. A biopsy confirmed clear cell odontogenic carcinoma, and the patient was treated with definitive surgery and radiation for localised disease. Unfortunately, the patient was found to have pulmonary metastases 3 months after initial management and was subsequently treated with a combination of cytotoxic chemotherapy and immunotherapy with a partial response. To our knowledge, this is the first case demonstrating the successful use of chemoimmunotherapy in metastatic clear cell odontogenic carcinoma.

References

Yancoskie A, Sreekantaiah C, Jacob J, Rosenberg A, Edelman M, Antonescu C . EWSR1 and ATF1 rearrangements in clear cell odontogenic carcinoma: presentation of a case. Oral Surg Oral Med Oral Pathol Oral Radiol. 2014; 118(4):e115-8. DOI: 10.1016/j.oooo.2014.02.004. View

Tajima S, Namiki I, Koda K . A clear cell variant of mucoepidermoid carcinoma harboring CRTC1-MAML2 fusion gene found in buccal mucosa: report of a case showing a large clear cell component and lacking typical epidermoid cells and intermediate cells. Med Mol Morphol. 2015; 50(2):117-121. DOI: 10.1007/s00795-015-0120-5. View

Rooney M, Korpics M, Turchan W, Callahan N, Koshy M, Spiotto M . Patterns of Care and Survival Outcomes for Odontogenic Cancers. Laryngoscope. 2020; 131(5):E1496-E1502. PMC: 8689582. DOI: 10.1002/lary.29173. View

Guastaldi F, Faquin W, Gootkind F, Hashemi S, August M, Iafrate A . Clear cell odontogenic carcinoma: a rare jaw tumor. A summary of 107 reported cases. Int J Oral Maxillofac Surg. 2019; 48(11):1405-1410. PMC: 7422703. DOI: 10.1016/j.ijom.2019.05.006. View

Motzer R, Tannir N, Mcdermott D, Frontera O, Melichar B, Choueiri T . Nivolumab plus Ipilimumab versus Sunitinib in Advanced Renal-Cell Carcinoma. N Engl J Med. 2018; 378(14):1277-1290. PMC: 5972549. DOI: 10.1056/NEJMoa1712126. View

Larkin J, Chiarion-Sileni V, Gonzalez R, Grob J, Rutkowski P, Lao C . Five-Year Survival with Combined Nivolumab and Ipilimumab in Advanced Melanoma. N Engl J Med. 2019; 381(16):1535-1546. DOI: 10.1056/NEJMoa1910836. View

Ebert Jr C, Dubin M, Hart C, Chalian A, Shockley W . Clear cell odontogenic carcinoma: a comprehensive analysis of treatment strategies. Head Neck. 2005; 27(6):536-42. DOI: 10.1002/hed.20181. View

Swain N, Dhariwal R, Ray J . Clear cell odontogenic carcinoma of maxilla: A case report and mini review. J Oral Maxillofac Pathol. 2013; 17(1):89-94. PMC: 3687196. DOI: 10.4103/0973-029X.110681. View

Loyola A, Cardoso S, de Faria P, Servato J, Paulo L, Eisenberg A . Clear cell odontogenic carcinoma: report of 7 new cases and systematic review of the current knowledge. Oral Surg Oral Med Oral Pathol Oral Radiol. 2015; 120(4):483-96. DOI: 10.1016/j.oooo.2015.06.005. View

10.

Tsai C, Kuo Y, Wu H, Ho C, Chen Y, Yang C . Adjuvant Radiotherapy Significantly Increases Neck Control and Survival in Early Oral Cancer Patients with Solitary Nodal Involvement: A National Cancer Registry Database Analysis. Cancers (Basel). 2021; 13(15). PMC: 8345217. DOI: 10.3390/cancers13153742. View

11.

Peraza Labrador A, Marin N, Valdez L, Valentina M, Sanchez K, Ibazetta K . Clear Cell Odontogenic Carcinoma a Systematic Review. Head Neck Pathol. 2021; 16(3):838-848. PMC: 9424403. DOI: 10.1007/s12105-021-01383-9. View

12.

Caudell J, Gillison M, Maghami E, Spencer S, Pfister D, Adkins D . NCCN Guidelines® Insights: Head and Neck Cancers, Version 1.2022. J Natl Compr Canc Netw. 2022; 20(3):224-234. DOI: 10.6004/jnccn.2022.0016. View

13.

Ferreira S, Faverani L, Dos Santos G, Martins E, Junior I . Clear cell odontogenic carcinoma of the mandible: a treatment strategy. J Appl Oral Sci. 2018; 26:e20160645. PMC: 5777409. DOI: 10.1590/1678-7757-2016-0645. View

14.

ONeill I . t(11;19) translocation and CRTC1-MAML2 fusion oncogene in mucoepidermoid carcinoma. Oral Oncol. 2008; 45(1):2-9. DOI: 10.1016/j.oraloncology.2008.03.012. View

15.

Sun Y, Liu H, Lv Y, Chen F, Hu Y, Yang K . Recurring Clear Cell Odontogenic Carcinoma Involving Vertebra and Pleura: A Case Report and Literature Review. Int J Surg Pathol. 2022; 31(2):203-212. DOI: 10.1177/10668969221098082. View

16.

Bellmunt J, de Wit R, Vaughn D, Fradet Y, Lee J, Fong L . Pembrolizumab as Second-Line Therapy for Advanced Urothelial Carcinoma. N Engl J Med. 2017; 376(11):1015-1026. PMC: 5635424. DOI: 10.1056/NEJMoa1613683. View

17.

Bilodeau E, Weinreb I, Antonescu C, Zhang L, Dacic S, Muller S . Clear cell odontogenic carcinomas show EWSR1 rearrangements: a novel finding and a biological link to salivary clear cell carcinomas. Am J Surg Pathol. 2013; 37(7):1001-5. DOI: 10.1097/PAS.0b013e31828a6727. View

18.

DCruz A, Vaish R, Kapre N, Dandekar M, Gupta S, Hawaldar R . Elective versus Therapeutic Neck Dissection in Node-Negative Oral Cancer. N Engl J Med. 2015; 373(6):521-9. DOI: 10.1056/NEJMoa1506007. View