{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,19]],"date-time":"2026-06-19T05:12:04Z","timestamp":1781845924505,"version":"3.54.5"},"reference-count":35,"publisher":"Springer Science and Business Media LLC","issue":"4","license":[{"start":{"date-parts":[[2021,2,22]],"date-time":"2021-02-22T00:00:00Z","timestamp":1613952000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.springer.com\/tdm"},{"start":{"date-parts":[[2021,2,22]],"date-time":"2021-02-22T00:00:00Z","timestamp":1613952000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/www.springer.com\/tdm"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Head and Neck Pathol"],"published-print":{"date-parts":[[2021,12]]},"DOI":"10.1007\/s12105-021-01302-y","type":"journal-article","created":{"date-parts":[[2021,2,22]],"date-time":"2021-02-22T16:05:10Z","timestamp":1614009910000},"page":"1391-1398","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":21,"title":["Clear Cell Odontogenic Carcinoma: First Report of Novel EWSR1\u2013CREM Fusion Gene in Case of Long-Term Misdiagnosis"],"prefix":"10.1007","volume":"15","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7996-9268","authenticated-orcid":false,"given":"Omar","family":"Breik","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"James","family":"Higginson","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Abdulla K.","family":"Al-Ajami","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Abubakar","family":"Mohamed","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tim","family":"Martin","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rasoul","family":"Amel-Kashipaz","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2021,2,22]]},"reference":[{"key":"1302_CR1","doi-asserted-by":"publisher","first-page":"363","DOI":"10.1016\/j.jormas.2017.08.005","volume":"118","author":"M Hadj Sa\u00efd","year":"2017","unstructured":"Hadj Sa\u00efd M, Ordioni U, Benat G, Gomez-Brouchet A, Chossegros C, Catherine JH. Clear cell odontogenic carcinoma. A review. J Stomatol Oral Maxillofac Surg. 2017;118:363\u201370.","journal-title":"J Stomatol Oral Maxillofac Surg."},{"key":"1302_CR2","doi-asserted-by":"publisher","first-page":"101","DOI":"10.1007\/s12105-011-0244-4","volume":"5","author":"EA Bilodeau","year":"2011","unstructured":"Bilodeau EA, Hoschar AP, Barnes EL, Hunt JL, Seethala RR. Clear cell carcinoma and clear cell odontogenic carcinoma: a comparative clinicopathologic and immunohistochemical study. Head Neck Pathol. 2011;5:101\u20137. https:\/\/doi.org\/10.1007\/s12105-011-0244-4.","journal-title":"Head Neck Pathol"},{"key":"1302_CR3","doi-asserted-by":"publisher","first-page":"115","DOI":"10.1002\/hed.2890080208","volume":"8","author":"LS Hansen","year":"1985","unstructured":"Hansen LS, Eversole LR, Green TL, Powell NB. Clear cell odontogenic tumor\u2014a new histologic variant with aggressive potential. Head Neck Surg. 1985;8:115\u201323. https:\/\/doi.org\/10.1002\/hed.2890080208.","journal-title":"Head Neck Surg"},{"key":"1302_CR4","doi-asserted-by":"publisher","first-page":"68","DOI":"10.1007\/s12105-017-0794-1","volume":"11","author":"JM Wright","year":"2017","unstructured":"Wright JM, Vered M. Update from the 4th Edition of the World Health Organization Classification of head and neck tumours: odontogenic and maxillofacial bone tumors. Head Neck Pathol. 2017;11:68\u201377. https:\/\/doi.org\/10.1007\/s12105-017-0794-1.","journal-title":"Head Neck Pathol"},{"key":"1302_CR5","doi-asserted-by":"publisher","first-page":"1405","DOI":"10.1016\/j.ijom.2019.05.006","volume":"48","author":"FPS Guastaldi","year":"2019","unstructured":"Guastaldi FPS, Faquin WC, Gootkind F, Hashemi S, August M, Iafrate AJ, et al. Clear cell odontogenic carcinoma: a rare jaw tumor. A summary of 107 reported cases. Int J Oral Maxillofac Surg. 2019;48:1405\u201310. https:\/\/doi.org\/10.1016\/j.ijom.2019.05.006.","journal-title":"Int J Oral Maxillofac Surg."},{"key":"1302_CR6","doi-asserted-by":"publisher","first-page":"483","DOI":"10.1016\/j.oooo.2015.06.005","volume":"120","author":"AM Loyola","year":"2015","unstructured":"Loyola AM, Cardoso SV, de Faria PR, Servato JPS, de Paulo LF, Eisenberg ALA, et al. 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;120:483\u201396.","journal-title":"Oral Surg Oral Med Oral Pathol Oral Radiol."},{"key":"1302_CR7","doi-asserted-by":"publisher","first-page":"1001","DOI":"10.1097\/PAS.0b013e31828a6727","volume":"37","author":"EA Bilodeau","year":"2013","unstructured":"Bilodeau EA, Weinreb I, Antonescu CR, Zhang L, Dacic S, Muller S, et al. Clear cell odontogenic carcinomas show EWSR1 rearrangements. Am J Surg Pathol. 2013;37:1001\u20135.","journal-title":"Am J Surg Pathol"},{"key":"1302_CR8","doi-asserted-by":"publisher","first-page":"609","DOI":"10.1038\/s41379-018-0175-7","volume":"32","author":"G Zhu","year":"2019","unstructured":"Zhu G, Benayed R, Ho C, Mullaney K, Sukhadia P, Rios K, et al. Diagnosis of known sarcoma fusions and novel fusion partners by targeted RNA sequencing with identification of a recurrent ACTB-FOSB fusion in pseudomyogenic hemangioendothelioma. Mod Pathol. 2019;32:609\u201320.","journal-title":"Mod Pathol"},{"key":"1302_CR9","doi-asserted-by":"publisher","first-page":"1622","DOI":"10.1097\/PAS.0000000000001331","volume":"43","author":"A Yoshida","year":"2019","unstructured":"Yoshida A, Wakai S, Ryo E, Miyata K, Miyazawa M, Yoshida KI, et al. Expanding the phenotypic spectrum of mesenchymal tumors harboring the EWSR1-CREM fusion. Am J Surg Pathol. 2019;43:1622\u201330.","journal-title":"Am J Surg Pathol"},{"key":"1302_CR10","doi-asserted-by":"publisher","first-page":"580","DOI":"10.1053\/joms.2003.50114","volume":"61","author":"M August","year":"2003","unstructured":"August M, Faquin W, Troulis M, Kaban L. Clear cell odontogenic carcinoma: evaluation of reported cases. J Oral Maxillofac Surg. 2003;61:580\u20136.","journal-title":"J Oral Maxillofac Surg"},{"key":"1302_CR11","first-page":"18","volume":"10","author":"A Jain","year":"2016","unstructured":"Jain A, Shetty DC, Juneja S, Narwal N. Molecular characterization of clear cell lesions of head and neck. J Clin Diagn Res. 2016;10:18\u201323.","journal-title":"J Clin Diagn Res."},{"key":"1302_CR12","doi-asserted-by":"publisher","first-page":"165","DOI":"10.1016\/j.oraloncology.2017.07.021","volume":"72","author":"MG Diniz","year":"2017","unstructured":"Diniz MG, Gomes CC, de Sousa SF, Xavier GM, Gomez RS. Oncogenic signalling pathways in benign odontogenic cysts and tumours. Oral Oncol. 2017;72:165\u201373.","journal-title":"Oral Oncol"},{"issue":"9","key":"1302_CR13","doi-asserted-by":"publisher","first-page":"1062","DOI":"10.1038\/modpathol.3800147","volume":"17","author":"L Nodit","year":"2004","unstructured":"Nodit L, Barnes L, Childers E, Finkelstein S, Swalsky P, Hunt J. Allelic loss of tumor suppressor genes in ameloblastic tumors. Mod Pathol. 2004;17(9):1062\u20137.","journal-title":"Mod Pathol."},{"key":"1302_CR14","doi-asserted-by":"publisher","first-page":"310","DOI":"10.1111\/j.1600-0714.1984.tb01429.x","volume":"13","author":"MK Basu","year":"1984","unstructured":"Basu MK, Matthews JB, Sear AJ, Bkowne RM. Calcifying epithelial odontogenic tumour: a case showing features of malignancy. J Oral Pathol Med. 1984;13:310\u20139. https:\/\/doi.org\/10.1111\/j.1600-0714.1984.tb01429.x.","journal-title":"J Oral Pathol Med"},{"key":"1302_CR15","doi-asserted-by":"publisher","first-page":"692","DOI":"10.1002\/hed.1097","volume":"23","author":"MJ Veness","year":"2001","unstructured":"Veness MJ, Morgan G, Collins AP, Walker DM. Calcifying epithelial odontogenic (Pindborg) tumor with malignant transformation and metastatic spread. Head Neck. 2001;23:692\u20136.","journal-title":"Head Neck"},{"key":"1302_CR16","doi-asserted-by":"publisher","first-page":"287","DOI":"10.1067\/moe.2002.121991","volume":"93","author":"YSL Cheng","year":"2002","unstructured":"Cheng YSL, Wright JM, Walstad WR, Finn MD. Calcifying epithelial odontogenic tumor showing microscopic features of potential malignant behavior. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2002;93:287\u201395.","journal-title":"Oral Surg Oral Med Oral Pathol Oral Radiol Endod"},{"key":"1302_CR17","doi-asserted-by":"publisher","first-page":"1770","DOI":"10.1097\/00005537-200410000-00018","volume":"114","author":"D Goldenberg","year":"2004","unstructured":"Goldenberg D, Sciubba J, Koch W, Tufano RP. Malignant odontogenic tumors: a 22-year experience. Laryngoscope. 2004;114:1770\u20134. https:\/\/doi.org\/10.1097\/00005537-200410000-00018.","journal-title":"Laryngoscope"},{"key":"1302_CR18","doi-asserted-by":"publisher","first-page":"76","DOI":"10.1016\/j.tripleo.2006.04.014","volume":"104","author":"K Kawano","year":"2007","unstructured":"Kawano K, Ono K, Yada N, Takahashi Y, Kashima K, Yokoyama S, et al. Malignant calcifying epithelial odontogenic tumor of the mandible: report of a case with pulmonary metastasis showing remarkable response to platinum derivatives. Oral Surge Oral Med Oral Pathol Oral Radiol Endodontol. 2007;104:76\u201381.","journal-title":"Oral Surge Oral Med Oral Pathol Oral Radiol Endodontol"},{"key":"1302_CR19","doi-asserted-by":"publisher","first-page":"1964","DOI":"10.1016\/j.joms.2010.02.017","volume":"68","author":"N Demian","year":"2010","unstructured":"Demian N, Harris RJ, Abramovitch K, Wilson JW, Vigneswaran N. Malignant transformation of calcifying epithelial odontogenic tumor is associated with the loss of p53 transcriptional activity: a case report with review of the literature. J Oral Maxillofac Surg. 2010;68:1964\u201373. https:\/\/doi.org\/10.1016\/j.joms.2010.02.017.","journal-title":"J Oral Maxillofac Surg"},{"key":"1302_CR20","first-page":"157","volume":"13","author":"Y Zhong","year":"2010","unstructured":"Zhong Y, Wang L, Li T, Chen XM. Calcifying epithelial odontogenic tumour showing malignant transformation: a case report and review of the literature. Chin J Dent Res. 2010;13:157\u201362.","journal-title":"Chin J Dent Res"},{"key":"1302_CR21","doi-asserted-by":"publisher","first-page":"1117","DOI":"10.1016\/j.jcms.2017.05.007","volume":"45","author":"BR Chrcanovic","year":"2017","unstructured":"Chrcanovic BR, Gomez RS. Calcifying epithelial odontogenic tumor: an updated analysis of 339 cases reported in the literature. J Cranio-Maxillofacial Surg. 2017;45:1117\u201323. https:\/\/doi.org\/10.1016\/j.jcms.2017.05.007.","journal-title":"J Cranio-Maxillofacial Surg"},{"key":"1302_CR22","doi-asserted-by":"publisher","first-page":"287","DOI":"10.1067\/moe.2002.121991","volume":"93","author":"Y-SL Cheng","year":"2002","unstructured":"Cheng Y-SL, Wright JM, Walstad WR, Finn MD. Calcifying epithelial odontogenic tumor showing microscopic features of potential malignant behavior. Oral Surg Oral Med Oral Pathol Oral Radiol Endodontol. 2002;93:287\u201395.","journal-title":"Oral Surg Oral Med Oral Pathol Oral Radiol Endodontol."},{"key":"1302_CR23","doi-asserted-by":"publisher","first-page":"190","DOI":"10.1016\/S1368-8375(02)00012-X","volume":"39","author":"M Kumar","year":"2003","unstructured":"Kumar M, Fasanmade A, William Barrett A, Mack G, Newman L, Hyde NC. Metastasising clear cell odontogenic carcinoma: a case report and review of the literature. Oral Oncol. 2003;39:190\u20134.","journal-title":"Oral Oncol"},{"key":"1302_CR24","doi-asserted-by":"publisher","first-page":"541","DOI":"10.1038\/nrc3087","volume":"11","author":"BS Taylor","year":"2011","unstructured":"Taylor BS, Barretina J, Maki RG, Antonescu CR, Singer S, Ladanyi M. Advances in sarcoma genomics and new therapeutic targets. Nat Rev Cancer. 2011;11:541\u201357.","journal-title":"Nat Rev Cancer"},{"key":"1302_CR25","doi-asserted-by":"publisher","first-page":"559","DOI":"10.1002\/gcc.20881","volume":"50","author":"CR Antonescu","year":"2011","unstructured":"Antonescu CR, Katabi N, Zhang L, Sung YS, Seethala RR, Jordan RC, et al. EWSR1-ATF1 fusion is a novel and consistent finding in hyalinizing clear-cell carcinoma of salivary gland. Genes Chromosom Cancer. 2011;50:559\u201370. https:\/\/doi.org\/10.1002\/gcc.20881.","journal-title":"Genes Chromosom Cancer"},{"key":"1302_CR26","doi-asserted-by":"publisher","first-page":"571","DOI":"10.1097\/PAS.0b013e3182772a15","volume":"37","author":"AA Shah","year":"2013","unstructured":"Shah AA, LeGallo RD, van Zante A, Frierson HF, Mills SE, Berean KW, et al. EWSR1 genetic rearrangements in salivary gland tumors. Am J Surg Pathol. 2013;37:571\u20138.","journal-title":"Am J Surg Pathol"},{"key":"1302_CR27","doi-asserted-by":"publisher","first-page":"e115","DOI":"10.1016\/j.oooo.2014.02.004","volume":"118","author":"AE Yancoskie","year":"2014","unstructured":"Yancoskie AE, Sreekantaiah C, Jacob J, Rosenberg A, Edelman M, Antonescu CR, et al. EWSR1 and ATF1 rearrangements in clear cell odontogenic carcinoma: presentation of a case. Oral Surg Oral Med Oral Pathol Oral Radiol. 2014;118:e115\u20138.","journal-title":"Oral Surg Oral Med Oral Pathol Oral Radiol"},{"key":"1302_CR28","doi-asserted-by":"publisher","first-page":"225","DOI":"10.1007\/s12105-018-0953-z","volume":"13","author":"R Vogels","year":"2019","unstructured":"Vogels R, Baumhoer D, van Gorp J, Eijkelenboom A, Verdijk M, van Cleef P, et al. Clear cell odontogenic carcinoma: occurrence of EWSR1-CREB1 as alternative fusion gene to EWSR1-ATF1. Head Neck Pathol. 2019;13:225\u201330. https:\/\/doi.org\/10.1007\/s12105-018-0953-z.","journal-title":"Head Neck Pathol"},{"key":"1302_CR29","doi-asserted-by":"publisher","first-page":"847","DOI":"10.1007\/s12105-019-01103-4","volume":"14","author":"T Santana","year":"2020","unstructured":"Santana T, de Andrade FL, de Sousa Melo MC, da Rocha GBL, Trierveiler M. Clear cell odontogenic carcinoma harboring the EWSR1\u2013ATF1 fusion gene: report of a rare case. Head Neck Pathol. 2020;14:847\u201351. https:\/\/doi.org\/10.1007\/s12105-019-01103-4.","journal-title":"Head Neck Pathol"},{"key":"1302_CR30","doi-asserted-by":"publisher","first-page":"217","DOI":"10.1007\/s12105-015-0635-z","volume":"10","author":"DT Ginat","year":"2016","unstructured":"Ginat DT, Villaflor V, Cipriani NA. Oral cavity clear cell odontogenic carcinoma. Head Neck Pathol. 2016;10:217\u201320.","journal-title":"Head Neck Pathol"},{"key":"1302_CR31","doi-asserted-by":"publisher","first-page":"e1","DOI":"10.1097\/PAS.0b013e31825485c5","volume":"36","author":"K Thway","year":"2012","unstructured":"Thway K, Fisher C. Tumors with EWSR1-CREB1 and EWSR1-ATF1 fusions. Am J Surg Pathol. 2012;36:e1\u201311.","journal-title":"Am J Surg Pathol"},{"issue":"4","key":"1302_CR32","doi-asserted-by":"publisher","first-page":"482","DOI":"10.1097\/PAS.0000000000000788","volume":"41","author":"YC Kao","year":"2017","unstructured":"Kao YC, Sung YS, Zhang L, Chen CL, Vaiyapuri S, Rosenblum MK, et al. EWSR1 fusions with CREB family transcription factors define a novel myxoid mesenchymal tumor with predilection for intracranial location. Am J Surg Pathol. 2017;41(4):482.","journal-title":"Am J Surg Pathol"},{"issue":"1","key":"1302_CR33","doi-asserted-by":"publisher","first-page":"821","DOI":"10.1146\/annurev.biochem.68.1.821","volume":"68","author":"AJ Shaywitz","year":"1999","unstructured":"Shaywitz AJ, Greenberg ME. CREB: a stimulus-induced transcription factor activated by a diverse array of extracellular signals. Annu Rev Biochem. 1999;68(1):821\u201361.","journal-title":"Annu Rev Biochem"},{"key":"1302_CR34","doi-asserted-by":"publisher","first-page":"1182","DOI":"10.1097\/PAS.0000000000001114","volume":"42","author":"E Chapman","year":"2018","unstructured":"Chapman E, Skalova A, Ptakova N, Martinek P, Goytain A, Tucker T, et al. Molecular profiling of hyalinizing clear cell carcinomas revealed a subset of tumors harboring a Novel EWSR1-CREM fusion: Report of 3 Cases. Am J Surg Pathol. 2018;42:1182\u20139.","journal-title":"Am J Surg Pathol"},{"key":"1302_CR35","doi-asserted-by":"publisher","first-page":"255","DOI":"10.1016\/j.humpath.2014.10.018","volume":"46","author":"EA Bilodeau","year":"2015","unstructured":"Bilodeau EA, Acquafondata M, Barnes EL, Seethala RR. A comparative analysis of LEF-1 in odontogenic and salivary tumors. Hum Pathol. 2015;46:255\u20139.","journal-title":"Hum Pathol"}],"container-title":["Head and Neck Pathology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s12105-021-01302-y.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1007\/s12105-021-01302-y\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s12105-021-01302-y.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,12,1]],"date-time":"2021-12-01T06:36:58Z","timestamp":1638340618000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1007\/s12105-021-01302-y"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,2,22]]},"references-count":35,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2021,12]]}},"alternative-id":["1302"],"URL":"https:\/\/doi.org\/10.1007\/s12105-021-01302-y","relation":{},"ISSN":["1936-0568"],"issn-type":[{"value":"1936-0568","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,2,22]]},"assertion":[{"value":"19 December 2020","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"5 February 2021","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"22 February 2021","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Compliance with Ethical Standards"}},{"value":"The authors have no conflicts of interest to declare.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Conflict of interest"}},{"value":"Patient consent to publish was provided by the patient. Ethics approval to publish this case report was provided by the Queen Elizabeth Hospital Ethics Committee (Approval number CARMS-00093).","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethical Approval"}}]}}