{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,5]],"date-time":"2026-10-05T22:53:14Z","timestamp":1791240794208,"version":"4.1.0"},"reference-count":9,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2026,10,5]],"date-time":"2026-10-05T00:00:00Z","timestamp":1791158400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2026,10,5]],"date-time":"2026-10-05T00:00:00Z","timestamp":1791158400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Gen Thorac Cardiovasc Surg Cases"],"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>\n                      Hyalinizing clear cell carcinoma (HCCC) is a rare salivary gland\u2013type tumor that occasionally arises in the lung. As it shows substantial morphological overlap with mucoepidermoid carcinoma (MEC), definitive diagnosis can be challenging. Therefore, molecular testing is essential, as MEC typically harbors\n                      <jats:italic>CRTC1\/3::MAML2<\/jats:italic>\n                      fusions, whereas HCCC is characterized by\n                      <jats:italic>EWSR1<\/jats:italic>\n                      fusions.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Case presentation<\/jats:title>\n                    <jats:p>\n                      A 60-year-old woman presented with persistent cough and hemoptysis. Imaging revealed a 3.0-cm solid nodule in the left lower lobe adjacent to the bronchus. She underwent robot-assisted left lower lobectomy with lymph node dissection. Histologically, the tumor consisted of uniform epithelioid cells with clear-to-pale eosinophilic cytoplasm arranged in nests and cords within prominent hyalinized stroma. Cytologic atypia was minimal. Immunohistochemically, tumor cells were positive for CK7, CK5\/6, and p40 and negative for TTF-1. Venous invasion was present without lymph node metastasis. Break-apart fluorescence in situ hybridization revealed rearrangements of\n                      <jats:italic>EWSR1<\/jats:italic>\n                      and\n                      <jats:italic>ATF1<\/jats:italic>\n                      , strongly supporting the presence of an\n                      <jats:italic>EWSR1::ATF1<\/jats:italic>\n                      fusion, whereas\n                      <jats:italic>MAML2<\/jats:italic>\n                      rearrangement was absent. The patient remains recurrence-free 1 year after surgery.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>\n                      Accurately distinguishing primary pulmonary HCCC, which is rare and may mimic MEC, is essential for appropriate pathological classification and prognostic assessment. It is also important for thoracic surgeons to consider HCCC during the differential diagnosis of centrally located or endobronchial pulmonary tumors, particularly when a definitive preoperative diagnosis is challenging. Integrating histopathology with molecular testing for\n                      <jats:italic>EWSR1<\/jats:italic>\n                      and\n                      <jats:italic>MAML2<\/jats:italic>\n                      rearrangements is critical for accurate diagnosis.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1186\/s44215-026-00280-3","type":"journal-article","created":{"date-parts":[[2026,10,5]],"date-time":"2026-10-05T09:35:01Z","timestamp":1791192901000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Primary pulmonary hyalinizing clear cell carcinoma harboring EWSR1::ATF1 fusion: a case report"],"prefix":"10.1186","volume":"5","author":[{"ORCID":"https:\/\/orcid.org\/0009-0000-7819-304X","authenticated-orcid":false,"given":"Takashi","family":"Teishikata","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mariko","family":"Hashiguchi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kozue","family":"Nakahara","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yusuke","family":"Okamoto","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Masafumi","family":"Hiratsuka","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Keiji","family":"Kamohara","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2026,10,5]]},"reference":[{"key":"280_CR1","doi-asserted-by":"publisher","first-page":"471","DOI":"10.1016\/j.humpath.2014.11.010","volume":"46","author":"JJ Garc\u00eda","year":"2015","unstructured":"Garc\u00eda JJ, Jin L, Jackson SB, Larsen BT, Lewis JE, Sukov WR, Roden AC. Primary pulmonary hyalinizing clear cell carcinoma of bronchial submucosal gland origin. Hum Pathol. 2015;46:471\u20135.","journal-title":"Hum Pathol"},{"key":"280_CR2","doi-asserted-by":"publisher","first-page":"1509132","DOI":"10.3389\/fonc.2024.1509132","volume":"14","author":"CH Jian","year":"2024","unstructured":"Jian CH, Luo S, Wang JJ. Primary pulmonary hyalinizing clear cell carcinoma with EWSR1 gene translocation: a case report. Front Oncol. 2024;14:1509132.","journal-title":"Front Oncol"},{"key":"280_CR3","doi-asserted-by":"publisher","first-page":"1175279","DOI":"10.3389\/fonc.2023.1175279","volume":"13","author":"YL Wu","year":"2023","unstructured":"Wu YL, Wu F, Cao MF, Lan Y, Du MS, Yu ST, Wang Y, Yan XC, Bian XW, Duan GJ. Primary pulmonary hyalinizing clear cell carcinoma with fusions of both EWSR1::CREM and IRF2::NTRK3: report of a case with an aggressive behavior. Front Oncol. 2023;13:1175279.","journal-title":"Front Oncol"},{"issue":"2","key":"280_CR4","doi-asserted-by":"publisher","first-page":"e11","DOI":"10.1097\/PAS.0000000000000980","volume":"42","author":"A Sk\u00e1lov\u00e1","year":"2018","unstructured":"Sk\u00e1lov\u00e1 A, Stenman G, Simpson RHW, Hellquist H, Slouka D, Svoboda T, et al. The role of molecular testing in the differential diagnosis of salivary gland carcinomas. Am J Surg Pathol. 2018;42(2):e11\u201327.","journal-title":"Am J Surg Pathol"},{"key":"280_CR5","doi-asserted-by":"publisher","first-page":"1479","DOI":"10.1038\/modpathol.2014.72","volume":"27","author":"AC Roden","year":"2014","unstructured":"Roden AC, Garc\u00eda JJ, Wehrs RN, Colby TV, Khoor A, Leslie KO, et al. Histopathologic, immunophenotypic and cytogenetic features of pulmonary mucoepidermoid carcinoma. Mod Pathol. 2014;27:1479\u201388.","journal-title":"Mod Pathol"},{"key":"280_CR6","doi-asserted-by":"publisher","first-page":"85","DOI":"10.1111\/cas.12039","volume":"104","author":"H Noda","year":"2013","unstructured":"Noda H, Okumura Y, Nakayama T, Miyabe S, Fujiyoshi Y, Hattori H, et al. Clinicopathological significance of MAML2 gene split in mucoepidermoid carcinoma. Cancer Sci. 2013;104:85\u201392.","journal-title":"Cancer Sci"},{"issue":"1","key":"280_CR7","doi-asserted-by":"publisher","first-page":"35","DOI":"10.1186\/s13000-022-01216-5","volume":"17","author":"Y Zhang","year":"2022","unstructured":"Zhang Y, Han W, Zhou J, Yong X. Primary lung hyalinizing clear cell carcinoma: a diagnostic challenge in biopsy. Diagn Pathol. 2022;17(1):35.","journal-title":"Diagn Pathol"},{"issue":"1","key":"280_CR8","doi-asserted-by":"publisher","first-page":"90","DOI":"10.1186\/s13000-023-01376-y","volume":"18","author":"Z Li","year":"2023","unstructured":"Li Z, Li W, Xue L. Primary pulmonary hyalinizing clear cell carcinoma with vocal-cord squamous cell carcinoma: a case report with systematic review. Diagn Pathol. 2023;18(1):90.","journal-title":"Diagn Pathol"},{"key":"280_CR9","doi-asserted-by":"publisher","first-page":"e230045","DOI":"10.1148\/rg.230045","volume":"43","author":"F Girvin","year":"2023","unstructured":"Girvin F, Phan A, Steinberger S, Shostak E, Bessich J, Zhou F, et al. Malignant and benign tracheobronchial neoplasms: comprehensive review with radiologic, bronchoscopic, and pathologic correlation. Radiographics. 2023;43:e230045.","journal-title":"Radiographics"}],"container-title":["General Thoracic and Cardiovascular Surgery Cases"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s44215-026-00280-3.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s44215-026-00280-3","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s44215-026-00280-3.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,10,5]],"date-time":"2026-10-05T22:09:06Z","timestamp":1791238146000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1186\/s44215-026-00280-3"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,10,5]]},"references-count":9,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2026,12]]}},"alternative-id":["280"],"URL":"https:\/\/doi.org\/10.1186\/s44215-026-00280-3","relation":{},"ISSN":["2731-6203"],"issn-type":[{"value":"2731-6203","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,10,5]]},"assertion":[{"value":"27 May 2026","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"27 September 2026","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"5 October 2026","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"The need for ethical review and approval was waived by the ethics committee of our hospital, as case reports are exempt from review according to institutional policy.","order":1,"name":"Ethics","label":"Ethics approval and consent to participate","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"The patient provided written informed consent for the publication of this case report in a scientific journal.","order":2,"name":"Ethics","label":"Consent for publication","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"The authors declare that they have no competing interests.","order":3,"name":"Ethics","label":"Competing interests","group":{"name":"EthicsHeading","label":"Declarations"}}],"article-number":"44"}}