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Primary ES of the thyroid is exceedingly rare and few cases have been documented to date. We describe the case of a 54-year old woman with a history of breast carcinoma in whom a unique hypermetabolic left thyroid nodule was identified during a follow-up PET-CT scan. An ultrasound examination showed a hypoechogenic nodule of 3.7\u00a0cm. A cytological diagnosis of poorly differentiated thyroid carcinoma was made, and a total thyroidectomy was performed. The surgical specimen revealed a poorly differentiated neoplasm composed of medium-sized cells with scant cytoplasm, expressing pancytokeratin, CD99 and NKX2.2 but lacking p63 and p40 expression. Molecular analysis revealed a <jats:italic>EWSR1-FLI1<\/jats:italic> fusion transcript supporting the diagnosis of a primary extra-osseous ES of the thyroid. The patient received adjuvant chemotherapy and has no evidence of recurrent disease.<\/jats:p>","DOI":"10.1007\/s12105-021-01365-x","type":"journal-article","created":{"date-parts":[[2021,7,26]],"date-time":"2021-07-26T16:04:35Z","timestamp":1627315475000},"page":"581-586","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":9,"title":["Primary Extra-Osseous Ewing Sarcoma of the Thyroid: A Case Report and Review of the Literature"],"prefix":"10.1007","volume":"16","author":[{"given":"Amanda H.","family":"Seipel","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hiba","family":"Mechahougui","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nicolas","family":"Mach","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fr\u00e9d\u00e9ric","family":"Triponez","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"William C.","family":"Faquin","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4966-1278","authenticated-orcid":false,"given":"Claudio","family":"De Vito","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2021,7,26]]},"reference":[{"key":"1365_CR1","doi-asserted-by":"publisher","first-page":"1429","DOI":"10.6004\/jnccn.2018.0089","volume":"16","author":"RI Haddad","year":"2018","unstructured":"Haddad RI, Nasr C, Bischoff L, et al. 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