{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,3]],"date-time":"2026-09-03T08:13:01Z","timestamp":1788423181754,"version":"build-2803163510"},"reference-count":17,"publisher":"Wiley","license":[{"start":{"date-parts":[[2026,9,3]],"date-time":"2026-09-03T00:00:00Z","timestamp":1788393600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2026,9,3]],"date-time":"2026-09-03T00:00:00Z","timestamp":1788393600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Diagnostic Cytopathology"],"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>Pleural fluid involvement by high\u2010grade endometrial stromal sarcoma (HG\u2010ESS) is exceedingly rare, with only one previously reported case. We report a case of ZC3H7B::BCOR fusion\u2010positive HG\u2010ESS metastatic to the pleural fluid in a 32\u2010year\u2010old female who presented with pelvic pain and a large uterine mass suspicious for leiomyosarcoma on MRI. Additional imaging showed pulmonary and vertebral lesions suspicious for metastases. A total abdominal hysterectomy with bilateral salpingo\u2010oophorectomy revealed HG\u2010ESS, with RNA sequencing confirming a ZC3H7B::BCOR fusion. Despite trials of chemoradiation, she developed worsening metastatic burden and eventually presented with a large pleural effusion. Pleural fluid cytology demonstrated three\u2010dimensional clusters of spindled to epithelioid tumor cells with high nuclear\u2010to\u2010cytoplasmic ratios and coarse chromatin, an architectural pattern not seen on the primary resection. Correlation with the patient's known primary uterine tumor and its immunophenotypic features helped establish the diagnosis of metastatic HG\u2010ESS. Malignant effusions secondary to sarcoma account for fewer than 6% of cytologic fluid samples. This case expands the limited cytologic literature on metastatic HG\u2010ESS involving pleural fluid and emphasizes that metastatic HG\u2010ESS may display cytomorphologic features that differ from the primary tumor, making correlation with the patient's clinical history and prior resection essential for accurate diagnosis.<\/jats:p>","DOI":"10.1002\/dc.70205","type":"journal-article","created":{"date-parts":[[2026,9,3]],"date-time":"2026-09-03T07:22:01Z","timestamp":1788420121000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Metastatic High\u2010Grade Endometrial Stromal Sarcoma With\n                    <scp>ZC3H7B<\/scp>\n                    ::\n                    <scp>BCOR<\/scp>\n                    Fusion in Pleural Fluid: A Case Report"],"prefix":"10.1002","author":[{"ORCID":"https:\/\/orcid.org\/0009-0009-2673-6917","authenticated-orcid":false,"given":"Daniel","family":"Lauffenburger","sequence":"first","affiliation":[{"name":"Department of Pathology and Laboratory Medicine University of North Carolina Hospitals  Chapel Hill North Carolina USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0002-4887-6821","authenticated-orcid":false,"given":"Jan F.","family":"Silverman","sequence":"additional","affiliation":[{"name":"Department of Pathology and Laboratory Medicine University of North Carolina Hospitals  Chapel Hill North Carolina USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,9,3]]},"reference":[{"key":"e_1_2_8_2_1","volume-title":"Female Genital Tumours","author":"WHO Classification of Tumours Editorial Board","year":"2020"},{"issue":"11","key":"e_1_2_8_3_1","doi-asserted-by":"crossref","first-page":"1893","DOI":"10.3390\/cancers17111893","article-title":"Endometrial Stromal Sarcoma: An Update","volume":"17","author":"Ricotta G.","year":"2025","journal-title":"Cancers (Basel)"},{"issue":"2","key":"e_1_2_8_4_1","doi-asserted-by":"crossref","DOI":"10.1097\/MD.0000000000028490","article-title":"Clinicopathological Characteristics and Treatment of Patients With High\u2010Grade Endometrial Stromal Sarcoma: A Retrospective Study of 40 Cases","volume":"101","author":"Bai H.","year":"2022","journal-title":"Medicine (Baltimore)"},{"issue":"9","key":"e_1_2_8_5_1","doi-asserted-by":"crossref","first-page":"1161","DOI":"10.1097\/PAS.0000000000000256","article-title":"High\u2010Grade Endometrial Stromal Sarcomas: A Clinicopathologic Study of a Group of Tumors With Heterogeneous Morphologic and Genetic Features","volume":"38","author":"Sciallis A. P.","year":"2014","journal-title":"American Journal of Surgical Pathology"},{"issue":"4","key":"e_1_2_8_6_1","doi-asserted-by":"crossref","first-page":"440","DOI":"10.1097\/00000478-200204000-00005","article-title":"Endometrial Stromal Sarcoma Metastatic to the Lung: A Detailed Analysis of 16 Patients","volume":"26","author":"Aubry M. C.","year":"2002","journal-title":"American Journal of Surgical Pathology"},{"issue":"1","key":"e_1_2_8_7_1","doi-asserted-by":"crossref","first-page":"82","DOI":"10.1177\/10668969221098093","article-title":"Metastatic High\u2010Grade Endometrial Stromal Sarcoma With BCOR Translocation to the Pleural Fluid: A Case Report","volume":"31","author":"Gonsalves C.","year":"2023","journal-title":"International Journal of Surgical Pathology"},{"issue":"4","key":"e_1_2_8_8_1","doi-asserted-by":"crossref","first-page":"674","DOI":"10.1038\/modpathol.2017.162","article-title":"ZC3H7B\u2010BCOR High\u2010Grade Endometrial Stromal Sarcomas: A Report of 17 Cases of a Newly Defined Entity","volume":"31","author":"Lewis N.","year":"2018","journal-title":"Modern Pathology"},{"issue":"3","key":"e_1_2_8_9_1","doi-asserted-by":"crossref","first-page":"335","DOI":"10.1097\/PAS.0000000000000993","article-title":"BCOR Internal Tandem Duplication in High\u2010Grade Uterine Sarcomas","volume":"42","author":"Mari\u00f1o\u2010Enr\u00edquez A.","year":"2018","journal-title":"American Journal of Surgical Pathology"},{"issue":"2","key":"e_1_2_8_10_1","doi-asserted-by":"crossref","first-page":"71","DOI":"10.1002\/(SICI)1097-0142(19980425)84:2<71::AID-CNCR1>3.0.CO;2-G","article-title":"Cytologic Features of Sarcomas in Fluids","volume":"84","author":"Abadi M. 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S.","year":"2015","journal-title":"Diagnostic Cytopathology"},{"issue":"4","key":"e_1_2_8_13_1","doi-asserted-by":"crossref","first-page":"274","DOI":"10.1002\/dc.21431","article-title":"High\u2010Grade Endometrial Stromal Sarcoma With Smooth Muscle and Skeletal Muscle Differentiation: Report of a Case With Cytomorphologic and Immunocytologic Analysis","volume":"39","author":"Ohta Y.","year":"2011","journal-title":"Diagnostic Cytopathology"},{"issue":"3","key":"e_1_2_8_14_1","doi-asserted-by":"crossref","first-page":"216","DOI":"10.1016\/j.jasc.2023.01.005","article-title":"Metastatic Sarcomas to Pleural Effusion: A 10\u2010Year Large Tertiary Care Center Experience With Emphasis on Clinical Features and Cytomorphologic Characteristics","volume":"12","author":"Saoud C.","year":"2023","journal-title":"Journal of the American Society of Cytopathology"},{"issue":"10","key":"e_1_2_8_15_1","doi-asserted-by":"crossref","first-page":"776","DOI":"10.1002\/cncy.22431","article-title":"Bone and Soft Tissue Sarcomas in Cerebrospinal Fluid and Effusion: A 20\u2010Year Review at Our Institution","volume":"129","author":"Sharma A.","year":"2021","journal-title":"Cancer Cytopathology"},{"issue":"1","key":"e_1_2_8_16_1","doi-asserted-by":"crossref","first-page":"57","DOI":"10.1002\/(SICI)1097-0142(19970225)81:1<57::AID-CNCR12>3.0.CO;2-B","article-title":"Effusion Cytology of Malignant Melanoma","volume":"81","author":"Beaty M. 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