{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,18]],"date-time":"2026-05-18T09:09:14Z","timestamp":1779095354997,"version":"3.51.4"},"reference-count":30,"publisher":"Springer Science and Business Media LLC","license":[{"start":{"date-parts":[[2026,5,18]],"date-time":"2026-05-18T00:00:00Z","timestamp":1779062400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.springernature.com\/gp\/researchers\/text-and-data-mining"},{"start":{"date-parts":[[2026,5,18]],"date-time":"2026-05-18T00:00:00Z","timestamp":1779062400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/www.springernature.com\/gp\/researchers\/text-and-data-mining"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Virchows Arch"],"DOI":"10.1007\/s00428-026-04583-2","type":"journal-article","created":{"date-parts":[[2026,5,18]],"date-time":"2026-05-18T08:37:04Z","timestamp":1779093424000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Two rare cases of FET::CREB epithelioid mesenchymal tumors, displaying atypical immunophenotype (myoepithelial-like), unraveled on next-generation sequencing"],"prefix":"10.1007","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-3509-4794","authenticated-orcid":false,"given":"Bharat","family":"Rekhi","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Khushi","family":"Naik","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Prachi","family":"Bapat","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Swapnil","family":"Rane","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sonali Susmita","family":"Nayak","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mukta","family":"Ramadwar","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Omshree","family":"Shetty","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2026,5,18]]},"reference":[{"issue":"7","key":"4583_CR1","doi-asserted-by":"publisher","first-page":"e1","DOI":"10.1097\/PAS.0b013e31825485c5","volume":"36","author":"K Thway","year":"2012","unstructured":"Thway K, Fisher C (2012) Tumors with EWSR1-CREB1 and EWSR1-ATF1 fusions: the current status. Am J Surg Pathol 36(7):e1\u2013e11","journal-title":"Am J Surg Pathol"},{"key":"4583_CR2","doi-asserted-by":"publisher","first-page":"1051","DOI":"10.1002\/gcc.20491","volume":"46","author":"CR Antonescu","year":"2007","unstructured":"Antonescu CR, Dal Cin P, Nafa K, Teot LA, Surti U, Fletcher CD, Ladanyi M (2007) EWSR1-CREB1 is the predominant gene fusion in angiomatoid fibrous histiocytoma. Genes Chromosomes Cancer 46:1051\u20131060","journal-title":"Genes Chromosomes Cancer"},{"key":"4583_CR3","doi-asserted-by":"publisher","first-page":"695","DOI":"10.1002\/gcc.22474","volume":"56","author":"B Dewaele","year":"2017","unstructured":"Dewaele B, Libbrecht L, Levy G, Brichard B, Vanspauwen V, Sciot R, Debiec-Rychter M (2017) A novel EWS-CREB3L3 gene fusion in a mesenteric sclerosing epithelioid fibrosarcoma. Genes Chromosomes Cancer 56:695\u2013699","journal-title":"Genes Chromosomes Cancer"},{"key":"4583_CR4","doi-asserted-by":"publisher","first-page":"218","DOI":"10.1002\/gcc.20037","volume":"40","author":"I Panagopoulos","year":"2004","unstructured":"Panagopoulos I, Storlazzi CT, Fletcher CD, Fletcher JA, Nascimento A, Domanski HA, Wejde J, Brosj\u00f6 O, Rydholm A, Isaksson M, Mandahl N, Mertens F (2004) The chimeric FUS\/CREB3l2 gene is specific for low-grade fibromyxoid sarcoma. Genes Chromosomes Cancer 40:218\u2013228","journal-title":"Genes Chromosomes Cancer"},{"key":"4583_CR5","doi-asserted-by":"publisher","first-page":"101","DOI":"10.1016\/0165-4608(91)90059-4","volume":"52","author":"JA Bridge","year":"1991","unstructured":"Bridge JA, Sreekantaiah C, Neff JR, Sandberg AA (1991) Cytogenetic findings in clear cell sarcoma of tendons and aponeuroses. Malignant melanoma of soft parts. Cancer Genet Cytogenet 52:101\u2013106","journal-title":"Cancer Genet Cytogenet"},{"key":"4583_CR6","doi-asserted-by":"publisher","first-page":"1722","DOI":"10.1097\/PAS.0b013e318227e4d2","volume":"35","author":"K Thway","year":"2011","unstructured":"Thway K, Nicholson AG, Lawson K, Gonzalez D, Rice A, Balzer B, Swansbury J, Min T, Thompson L, Adu-Poku K, Campbell A, Fisher C (2011) Primary pulmonary myxoid sarcoma with EWSR1-CREB1 fusion: a new tumor entity. Am J Surg Pathol 35:1722\u20131732","journal-title":"Am J Surg Pathol"},{"key":"4583_CR7","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, Antonescu CR (2017) EWSR1 fusions with CREB family transcription factors define a novel myxoid mesenchymal tumor with predilection for intracranial location. Am J Surg Pathol 41:482\u2013490","journal-title":"Am J Surg Pathol"},{"key":"4583_CR8","doi-asserted-by":"publisher","DOI":"10.1111\/bpa.12918","volume":"31","author":"EA Sloan","year":"2021","unstructured":"Sloan EA, Chiang J, Villanueva-Meyer JE, Alexandrescu S, Eschbacher JM, Wang W, Mafra M, Ud Din N, Carr-Boyd E, Watson M, Punsoni M, Oviedo A, Gilani A, Kleinschmidt-DeMasters BK, Coss DJ, Lopes MB, Raffel C, Berger MS, Chang SM, Reddy A, Ramani B, Ferris SP, Lee JC, Hofmann JW, Cho SJ, Horvai AE, Pekmezci M, Tihan T, Bollen AW, Rodriguez FJ, Ellison DW, Perry A, Solomon DA (2021) Intracranial mesenchymal tumor with FET-CREB fusion-a unifying diagnosis for the spectrum of intracranial myxoid mesenchymal tumors and angiomatoid fibrous histiocytoma-like neoplasms. Brain Pathol 31:e12918","journal-title":"Brain Pathol"},{"issue":"7","key":"4583_CR9","doi-asserted-by":"publisher","DOI":"10.1016\/j.modpat.2023.100160","volume":"36","author":"V Ulici","year":"2023","unstructured":"Ulici V, Hornick JL, Davis JL, Mehrotra S, Meis JM, Halling KC, Fletcher CDM, Kao E, Folpe AL (2023) Extraenteric malignant gastrointestinal neuroectodermal tumor-a clinicopathologic and molecular genetic study of 11 cases. Mod Pathol 36(7):100160. https:\/\/doi.org\/10.1016\/j.modpat.2023.100160","journal-title":"Mod Pathol"},{"key":"4583_CR10","doi-asserted-by":"publisher","first-page":"504","DOI":"10.1097\/PAS.0000000000002022","volume":"47","author":"AM Acosta","year":"2023","unstructured":"Acosta AM, Bridge JA, Dal Cin PS, Sholl LM, Cornejo KM, Fletcher CDM, Ulbright TM (2023) Inflammatory and nested testicular sex cord tumor: a novel neoplasm with aggressive clinical behavior and frequent EWSR1::ATF1 gene fusions. Am J Surg Pathol 47:504\u2013517","journal-title":"Am J Surg Pathol"},{"key":"4583_CR11","doi-asserted-by":"crossref","unstructured":"Trecourt A, Macagno N, Ngo C, Philip CA, Lopez J, Ferreira J, Alves-Vale C, Ray-Coquard I, Genestie C, Agaimy A, Devouassoux-Shisheboran M (2023) CREB fusion-associated epithelioid mesenchymal neoplasms of the female adnexa: three cases documenting a novel location of an emerging entity and further highlighting an ambiguous misleading immunophenotype. Virchows Arch 2023; 482:967\u2013974","DOI":"10.1007\/s00428-023-03546-1"},{"key":"4583_CR12","doi-asserted-by":"publisher","first-page":"2233","DOI":"10.1038\/s41379-020-0646-5","volume":"33","author":"P Argani","year":"2020","unstructured":"Argani P, Harvey I, Nielsen GP, Takano A, Suurmeijer AJH, Voltaggio L, Zhang L, Sung YS, Stenzinger A, Mechtersheimer G, Dickson BC, Antonescu CR (2020) EWSR1\/FUS-CREB fusions define a distinctive malignant epithelioid neoplasm with predilection for mesothelial-lined cavities. Mod Pathol 33:2233\u20132243","journal-title":"Mod Pathol"},{"key":"4583_CR13","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, Motoi T, Ogawa C, Iwata S, Kobayashi E, Watanabe SI, Kawai A, Mori T (2019) Expanding the phenotypic spectrum of mesenchymal tumors harboring the EWSR1-CREM fusion. Am J Surg Pathol 43:1622\u20131630","journal-title":"Am J Surg Pathol"},{"key":"4583_CR14","doi-asserted-by":"crossref","unstructured":"Shibayama T, Shimoi T, Mori T, Noguchi E, Honma Y, Hijioka S, Yoshida M, Ogawa C, Yonemori K, Yatabe Y, Yoshida A (2022) Cytokeratin-positive Malignant Tumor in the Abdomen With EWSR1\/FUS-CREB Fusion: A Clinicopathologic Study of 8 Cases. Am J Surg Pathol 2022; 46:134\u2013146","DOI":"10.1097\/PAS.0000000000001742"},{"key":"4583_CR15","doi-asserted-by":"publisher","first-page":"995","DOI":"10.1007\/s00428-024-03879-5","volume":"485","author":"B Rekhi","year":"2024","unstructured":"Rekhi B, Dermawan JK, Fritchie KJ, Zimpfer A, Mohammad TM, Ali FS, Nandy K, Zou Y, Stoehr R, Agaimy A (2024) EWSR1::ATF1 fusions characterize a group of extra-abdominal epithelioid and round cell mesenchymal neoplasms, phenotypically overlapping with sclerosing epithelioid fibrosarcomas, and intra-abdominal FET::CREB fusion neoplasms. Virchows Arch 485:995\u20131005","journal-title":"Virchows Arch"},{"key":"4583_CR16","doi-asserted-by":"publisher","unstructured":"Rusidz\u00e9 M, Poumeaud F, Akiki B, Valentin T, Ferron G, Ducassou A, Pissaloux D, Evrard S, Brousset P, Le Guellec S, Rochaix P (2025) EWSR1::CREM rearranged intra-abdominal malignant epithelioid neoplasm: two new cases of an emerging entity with clinicopathological characteristics and histological pitfalls. Virchows Arch Jan31. https:\/\/doi.org\/10.1007\/s00428-025-04034-4. Epub ahead of print","DOI":"10.1007\/s00428-025-04034-4"},{"key":"4583_CR17","doi-asserted-by":"publisher","first-page":"1183","DOI":"10.1097\/00000478-200309000-00001","volume":"27","author":"JL Hornick","year":"2003","unstructured":"Hornick JL, Fletcher CD (2003) Myoepithelial tumors of soft tissue: a clinicopathologic and immunohistochemical study of 101 cases with evaluation of prognostic parameters. Am J Surg Pathol 27:1183\u20131196","journal-title":"Am J Surg Pathol"},{"key":"4583_CR18","doi-asserted-by":"publisher","first-page":"816","DOI":"10.1097\/PAS.0000000000000172","volume":"38","author":"IM Schaefer","year":"2014","unstructured":"Schaefer IM, Fletcher CD (2014) Myxoid variant of so-called angiomatoid malignant fibrous histiocytoma: clinicopathologic characterization in a series of 21 cases. Am J Surg Pathol 38:816\u2013823","journal-title":"Am J Surg Pathol"},{"key":"4583_CR19","doi-asserted-by":"publisher","first-page":"587","DOI":"10.1007\/s00428-025-04199-y","volume":"487","author":"A Agaimy","year":"2025","unstructured":"Agaimy A, Molligan J, Alruwaii FI, Antonescu CR, Demicco EG, Dickson BC, Gross J, Michal M, Perry K, T\u00f6gel L, Stoehr R, Ud Din N, Folpe AL (2025) Angiomatoid fibrous histiocytoma occurring at distal\/acral extremity sites: clinicopathological and molecular study of 26 cases highlighting frequent myxoid histology and site-dependent genotypic variation. Virchows Arch 487:587\u2013595","journal-title":"Virchows Arch"},{"key":"4583_CR20","doi-asserted-by":"publisher","first-page":"826","DOI":"10.1097\/PAS.0000000000000398","volume":"39","author":"M Miettinen","year":"2015","unstructured":"Miettinen M, McCue PA, Sarlomo-Rikala M, Biernat W, Czapiewski P, Kopczynski J, Thompson LD, Lasota J, Wang Z, Fetsch JF (2015) Sox10\u2013a marker for not only schwannian and melanocytic neoplasms but also myoepithelial cell tumors of soft tissue: a systematic analysis of 5134 tumors. Am J Surg Pathol 39:826\u2013835","journal-title":"Am J Surg Pathol"},{"key":"4583_CR21","doi-asserted-by":"crossref","unstructured":"Perret R, Escuriol J, Velasco V, Mayeur L, Soubeyran I, Delfour C et al (2020) NFATc2-rearranged sarcomas: clinicopathologic, molecular, and cytogenetic study of 7 cases with evidence of AGGRECAN as a novel diagnostic marker. Mod Pathol 2020;33:1930\u20131944","DOI":"10.1038\/s41379-020-0542-z"},{"key":"4583_CR22","doi-asserted-by":"publisher","first-page":"1275","DOI":"10.1177\/10668969241228294","volume":"32","author":"K Kosemehmetoglu","year":"2024","unstructured":"Kosemehmetoglu K, Rekhi B, Erdem ZB, Yildiz AE, Comunoglu N (2024) Clinicopathological features of three rare EWSR1::NFATC2 sarcomas of bone and soft tissues. Int J Surg Pathol 32:1275\u20131285","journal-title":"Int J Surg Pathol"},{"issue":"7","key":"4583_CR23","doi-asserted-by":"publisher","DOI":"10.1016\/j.modpat.2023.100160","volume":"36","author":"V Ulici","year":"2023","unstructured":"Ulici V, Hornick JL, Davis JL, Mehrotra S, Meis JM, Halling KC, Fletcher CDM, Kao E, Folpe AL (2023) Extraenteric malignant gastrointestinal neuroectodermal tumor-a clinicopathologic and molecular genetic study of 11 cases. Mod Pathol 36(7):100160. https:\/\/doi.org\/10.1016\/j.modpat.2023.100160","journal-title":"Mod Pathol"},{"key":"4583_CR24","doi-asserted-by":"publisher","first-page":"131","DOI":"10.1007\/s00428-022-03274-y","volume":"481","author":"CT Kuo","year":"2022","unstructured":"Kuo CT, Kao YC, Huang HY, Hsiao CH, Lee JC (2022) Malignant gastrointestinal neuroectodermal tumor in head and neck: two challenging cases with diverse morphology and different considerations for differential diagnosis. Virchows Arch 481:131\u2013136","journal-title":"Virchows Arch"},{"key":"4583_CR25","doi-asserted-by":"publisher","first-page":"1114","DOI":"10.1002\/gcc.20819","volume":"49","author":"CR Antonescu","year":"2010","unstructured":"Antonescu CR, Zhang L, Chang NE, Pawel BR, Travis W, Katabi N, Edelman M, Rosenberg AE, Nielsen GP, Dal Cin P, Fletcher CD (2010) EWSR1-POU5F1 fusion in soft tissue myoepithelial tumors. A molecular analysis of sixty-six cases, including soft tissue, bone, and visceral lesions, showing common involvement of the EWSR1 gene. Genes Chromosomes Cancer 49:1114\u20131124","journal-title":"Genes Chromosomes Cancer"},{"key":"4583_CR26","doi-asserted-by":"publisher","first-page":"687","DOI":"10.1007\/s00428-012-1335-7","volume":"461","author":"B Rekhi","year":"2012","unstructured":"Rekhi B, Sable M, Jambhekar NA (2012) Histopathological, immunohistochemical and molecular spectrum of myoepithelial tumours of soft tissues. Virchows Arch 461:687\u2013697","journal-title":"Virchows Arch"},{"key":"4583_CR27","doi-asserted-by":"publisher","first-page":"32","DOI":"10.1007\/s12105-015-0618-0","volume":"9","author":"VY Jo","year":"2015","unstructured":"Jo VY, Fletcher CD (2015) Myoepithelial neoplasms of soft tissue: an updated review of the clinicopathologic, immunophenotypic, and genetic features. Head Neck Pathol 9:32\u201338","journal-title":"Head Neck Pathol"},{"key":"4583_CR28","doi-asserted-by":"crossref","unstructured":"Flucke U, Mentzel T, Verdijk MA, Slootweg PJ, Creytens DH, Suurmeijer AJ, Tops BB (2012) EWSR1-ATF1 chimeric transcript in a myoepithelial tumor of soft tissue: a case report. Hum Pathol 2012 43:764\u2013768","DOI":"10.1016\/j.humpath.2011.08.004"},{"key":"4583_CR29","doi-asserted-by":"publisher","first-page":"258","DOI":"10.1177\/1093526621998869","volume":"24","author":"BD Leckey Jr","year":"2021","unstructured":"Leckey BD Jr, John I, Reyes-M\u00fagica M, Naous R (2021) EWSR1-ATF1 fusion in a myoepithelial carcinoma of soft tissue with small round cell morphology: a potential diagnostic pitfall. Pediatr Dev Pathol 24:258\u2013263","journal-title":"Pediatr Dev Pathol"},{"key":"4583_CR30","doi-asserted-by":"publisher","first-page":"628","DOI":"10.1158\/1078-0432.CCR-25-2426","volume":"32","author":"M Michal","year":"2026","unstructured":"Michal M, Malik F, Mansour B, Hattery T, Dehner CA, Mart\u00ednek P, H\u00e1jkov\u00e1 V, Van\u011b\u010dek T, Chrisinger JSA, Machado I, Klub\u00ed\u010dkov\u00e1 N, Sumathi VP, Ng T, Warmke L, Oon ML, Petersson F, Argani P, Gross JM, Michal M, Antonescu CR, Dermawan JK (2026) FET-Rearranged myoepithelial tumors are clinically heterogeneous and epigenetically distinct from PLAG1-rearranged adnexal and salivary gland myoepithelial tumors. Clin Cancer Res 32:628\u2013644","journal-title":"Clin Cancer Res"}],"container-title":["Virchows Archiv"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s00428-026-04583-2.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1007\/s00428-026-04583-2","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s00428-026-04583-2.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,5,18]],"date-time":"2026-05-18T08:37:07Z","timestamp":1779093427000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1007\/s00428-026-04583-2"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,5,18]]},"references-count":30,"alternative-id":["4583"],"URL":"https:\/\/doi.org\/10.1007\/s00428-026-04583-2","relation":{},"ISSN":["0945-6317","1432-2307"],"issn-type":[{"value":"0945-6317","type":"print"},{"value":"1432-2307","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,5,18]]},"assertion":[{"value":"24 February 2026","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"2 May 2026","order":2,"name":"revised","label":"Revised","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"9 May 2026","order":3,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"18 May 2026","order":4,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"The manuscript adheres and complies with the ethical standards.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Compliance with ethical standards"}},{"value":"None.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Conflict of interest"}}]}}