{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,15]],"date-time":"2026-10-15T04:23:40Z","timestamp":1792038220190,"version":"4.4.0"},"reference-count":31,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2019,4,22]],"date-time":"2019-04-22T00:00:00Z","timestamp":1555891200000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Diagn Pathol"],"published-print":{"date-parts":[[2019,12]]},"DOI":"10.1186\/s13000-019-0807-3","type":"journal-article","created":{"date-parts":[[2019,4,22]],"date-time":"2019-04-22T14:02:35Z","timestamp":1555941755000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":12,"title":["Metastatic low-grade endometrial stromal sarcoma of uterus presenting as a primary pancreatic tumor: case presentation and literature review"],"prefix":"10.1186","volume":"14","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-0806-0924","authenticated-orcid":false,"given":"Aoife J.","family":"McCarthy","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Blaise A.","family":"Clarke","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ian","family":"McGilvray","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Brendan C.","family":"Dickson","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Korosh","family":"Khalili","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Runjan","family":"Chetty","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2019,4,22]]},"reference":[{"issue":"6","key":"807_CR1","doi-asserted-by":"publisher","first-page":"527","DOI":"10.1007\/s00428-004-0987-3","volume":"444","author":"NV Adsay","year":"2004","unstructured":"Adsay NV, Andea A, Basturk O, et al. Secondary tumors of the pancreas: an analysis of a surgical and autopsy database and review of the literature. Virchows Arch. 2004;444(6):527\u201335.","journal-title":"Virchows Arch"},{"issue":"7","key":"807_CR2","doi-asserted-by":"publisher","first-page":"675","DOI":"10.1007\/BF02574484","volume":"9","author":"SP Hiotis","year":"2002","unstructured":"Hiotis SP, Klimstra DS, Conlon KC, et al. Results after pancreatic resection for metastatic lesions. Ann Surg Oncol. 2002;9(7):675\u20139.","journal-title":"Ann Surg Oncol"},{"issue":"4","key":"807_CR3","doi-asserted-by":"publisher","first-page":"536","DOI":"10.5858\/arpa.2017-0241-RA","volume":"142","author":"RP Masand","year":"2018","unstructured":"Masand RP. Unusual presentations of gynecologic tumors: primary, Extrauterine, low-grade Endometrioid stromal sarcoma. Arch Pathol Lab Med. 2018;142(4):536\u201341.","journal-title":"Arch Pathol Lab Med"},{"key":"807_CR4","series-title":"World Health Organization Classification of Tumours","volume-title":"Classification of Tumours of Female Reproductive Organs","author":"RJ Kurman","year":"2014","unstructured":"Kurman RJ, Carcangiu ML, Herrington S, Young RH, WHO. Classification of Tumours of Female Reproductive Organs, World Health Organization Classification of Tumours, vol. 6. 4th ed. Lyon: IARC Press; 2014.","edition":"4"},{"issue":"2","key":"807_CR5","doi-asserted-by":"publisher","first-page":"162","DOI":"10.1016\/j.pathol.2017.11.086","volume":"50","author":"L Hoang","year":"2018","unstructured":"Hoang L, Chiang S, Lee CH. Endometrial stromal sarcomas and related neoplasms: new developments and diagnostic considerations. Pathology. 2018;50(2):162\u201377.","journal-title":"Pathology"},{"issue":"3","key":"807_CR6","doi-asserted-by":"publisher","first-page":"630","DOI":"10.1016\/j.ygyno.2007.01.031","volume":"105","author":"CA Leath 3rd","year":"2007","unstructured":"Leath CA 3rd, Huh WK, Hyde J Jr, et al. A multi-institutional review of outcomes of endometrial stromal sarcoma. Gynecol Oncol. 2007;105(3):630\u20134.","journal-title":"Gynecol Oncol"},{"issue":"2","key":"807_CR7","doi-asserted-by":"publisher","first-page":"160","DOI":"10.1006\/gyno.2001.6152","volume":"81","author":"K Bodner","year":"2001","unstructured":"Bodner K, Bodner-Adler B, Obermair A, et al. Prognostic parameters in endometrial stromal sarcoma: a clinicopathologic study in 31 patients. Gynecol Oncol. 2001;81(2):160\u20135.","journal-title":"Gynecol Oncol"},{"issue":"3","key":"807_CR8","doi-asserted-by":"publisher","first-page":"654","DOI":"10.1016\/j.ygyno.2013.12.032","volume":"132","author":"H Bai","year":"2014","unstructured":"Bai H, Yang J, Cao D, et al. Ovary and uterus-sparing procedures for low-grade endometrial stromal sarcoma: a retrospective study of 153 cases. Gynecol Oncol. 2014;132(3):654\u201360.","journal-title":"Gynecol Oncol"},{"issue":"9","key":"807_CR9","doi-asserted-by":"publisher","first-page":"1364","DOI":"10.1097\/PAS.0b013e3182262743","volume":"35","author":"S Chiang","year":"2011","unstructured":"Chiang S, Ali R, Melnyk N, et al. Frequency of known gene rearrangements in endometrial stromal tumors. Am J Surg Pathol. 2011;35(9):1364\u201372.","journal-title":"Am J Surg Pathol"},{"issue":"11","key":"807_CR10","doi-asserted-by":"publisher","first-page":"1635","DOI":"10.1097\/PAS.0000000000000083","volume":"37","author":"RP Masand","year":"2013","unstructured":"Masand RP, Euscher ED, Deavers MT, et al. Endometrioid stromal sarcoma: a clinicopathologic study of 63 cases. Am J Surg Pathol. 2013;37(11):1635\u201347.","journal-title":"Am J Surg Pathol"},{"issue":"5","key":"807_CR11","doi-asserted-by":"publisher","first-page":"1143","DOI":"10.1002\/1097-0142(19840301)53:5<1143::AID-CNCR2820530521>3.0.CO;2-F","volume":"53","author":"RH Young","year":"1984","unstructured":"Young RH, Prat J, Scully RE. Endometrioid stromal sarcomas of the ovary. A clinicopathologic analysis of 23 cases. Cancer. 1984;53(5):1143\u201355.","journal-title":"Cancer"},{"issue":"2","key":"807_CR12","doi-asserted-by":"publisher","first-page":"224","DOI":"10.1097\/00000478-200402000-00010","volume":"28","author":"HY Huang","year":"2004","unstructured":"Huang HY, Ladanyi M, Soslow RA. Molecular detection of JAZF1-JJAZ1 gene fusion in endometrial stromal neoplasms with classic and variant histology: evidence for genetic heterogeneity. Am J Surg Pathol. 2004;28(2):224\u201332.","journal-title":"Am J Surg Pathol"},{"issue":"8","key":"807_CR13","doi-asserted-by":"publisher","first-page":"1228","DOI":"10.1097\/PAS.0b013e31816a3b42","volume":"32","author":"S Kurihara","year":"2008","unstructured":"Kurihara S, Oda Y, Ohishi Y, et al. Endometrial stromal sarcomas and related high-grade sarcomas: immunohistochemical and molecular genetic study of 31 cases. Am J Surg Pathol. 2008;32(8):1228\u201338.","journal-title":"Am J Surg Pathol"},{"issue":"2","key":"807_CR14","doi-asserted-by":"publisher","first-page":"119","DOI":"10.1016\/S0165-4608(03)00025-6","volume":"144","author":"F Micci","year":"2003","unstructured":"Micci F, Walter CU, Teixeira MR, et al. Cytogenetic and molecular genetic analyses of endometrial stromal sarcoma: nonrandom involvement of chromosome arms 6p and 7p and confirmation of JAZF1\/JJAZ1 gene fusion in t(7;17). Cancer Genet Cytogenet. 2003;144(2):119\u201324.","journal-title":"Cancer Genet Cytogenet"},{"issue":"1","key":"807_CR15","doi-asserted-by":"publisher","first-page":"65","DOI":"10.1097\/01.pas.0000213327.86992.d1","volume":"31","author":"MR Nucci","year":"2007","unstructured":"Nucci MR, Harburger D, Koontz J, et al. Molecular analysis of the JAZF1-JJAZ1 gene fusion by RT-PCR and fluorescence in situ hybridization in endometrial stromal neoplasms. Am J Surg Pathol. 2007;31(1):65\u201370.","journal-title":"Am J Surg Pathol"},{"issue":"8","key":"807_CR16","doi-asserted-by":"publisher","first-page":"1277","DOI":"10.1097\/PAS.0b013e318031f012","volume":"31","author":"E Oliva","year":"2007","unstructured":"Oliva E, de Leval L, Soslow RA, et al. High frequency of JAZF1-JJAZ1 gene fusion in endometrial stromal tumors with smooth muscle differentiation by interphase FISH detection. Am J Surg Pathol. 2007;31(8):1277\u201384.","journal-title":"Am J Surg Pathol"},{"issue":"4","key":"807_CR17","doi-asserted-by":"publisher","first-page":"514","DOI":"10.1097\/PAS.0b013e318272c612","volume":"37","author":"E D'Angelo","year":"2013","unstructured":"D'Angelo E, Ali RH, Espinosa I, et al. Endometrial stromal sarcomas with sex cord differentiation are associated with PHF1 rearrangement. Am J Surg Pathol. 2013;37(4):514\u201321.","journal-title":"Am J Surg Pathol"},{"issue":"6","key":"807_CR18","doi-asserted-by":"publisher","first-page":"780","DOI":"10.1111\/j.1365-2559.2008.03022.x","volume":"52","author":"S Regauer","year":"2008","unstructured":"Regauer S, Emberger W, Reich O, et al. Cytogenetic analyses of two new cases of endometrial stromal sarcoma--non-random reciprocal translocation t(10;17)(q22;p13) correlates with fibrous ESS. Histopathology. 2008;52(6):780\u20133.","journal-title":"Histopathology"},{"issue":"1","key":"807_CR19","doi-asserted-by":"publisher","first-page":"107","DOI":"10.1158\/0008-5472.CAN-05-2485","volume":"66","author":"F Micci","year":"2006","unstructured":"Micci F, Panagopoulos I, Bjerkehagen B, et al. Consistent rearrangement of chromosomal band 6p21 with generation of fusion genes JAZF1\/PHF1 and EPC1\/PHF1 in endometrial stromal sarcoma. Cancer Res. 2006;66(1):107\u201312.","journal-title":"Cancer Res"},{"issue":"6","key":"807_CR20","doi-asserted-by":"publisher","first-page":"e39354","DOI":"10.1371\/journal.pone.0039354","volume":"7","author":"I Panagopoulos","year":"2012","unstructured":"Panagopoulos I, Micci F, Thorsen J, et al. Novel fusion of MYST\/Esa1-associated factor 6 and PHF1 in endometrial stromal sarcoma. PLoS One. 2012;7(6):e39354.","journal-title":"PLoS One"},{"issue":"12","key":"807_CR21","doi-asserted-by":"publisher","first-page":"841","DOI":"10.1002\/gcc.22485","volume":"56","author":"F Micci","year":"2017","unstructured":"Micci F, Brunetti M, Dal Cin P, et al. Fusion of the genes BRD8 and PHF1 in endometrial stromal sarcoma. Genes Chromosom Cancer. 2017;56(12):841\u20135.","journal-title":"Genes Chromosom Cancer"},{"issue":"5","key":"807_CR22","doi-asserted-by":"publisher","first-page":"1112","DOI":"10.1002\/ijc.28440","volume":"134","author":"B Dewaele","year":"2014","unstructured":"Dewaele B, Przybyl J, Quattrone A, et al. Identification of a novel, recurrent MBTD1-CXorf67 fusion in low-grade endometrial stromal sarcoma. Int J Cancer. 2014;134(5):1112\u201322.","journal-title":"Int J Cancer"},{"issue":"27","key":"807_CR23","doi-asserted-by":"publisher","first-page":"19203","DOI":"10.18632\/oncotarget.24969","volume":"9","author":"M Brunetti","year":"2018","unstructured":"Brunetti M, Gorunova L, Davidson B, et al. Identification of an EPC2-PHF1 fusion transcript in low-grade endometrial stromal sarcoma. Oncotarget. 2018;9(27):19203\u20138.","journal-title":"Oncotarget."},{"issue":"11","key":"807_CR24","doi-asserted-by":"publisher","first-page":"598","DOI":"10.1002\/gcc.22649","volume":"57","author":"BC Dickson","year":"2018","unstructured":"Dickson BC, Lum A. Swanson, et al. novel EPC1 gene fusions in endometrial stromal sarcoma. Genes Chromosom Cancer. 2018;57(11):598\u2013603.","journal-title":"Genes Chromosom Cancer"},{"issue":"7","key":"807_CR25","doi-asserted-by":"publisher","first-page":"939","DOI":"10.1016\/j.humpath.2010.11.001","volume":"42","author":"C Amador-Ortiz","year":"2011","unstructured":"Amador-Ortiz C, Roma AA, Huettner PC, et al. JAZF1 and JJAZ1 gene fusion in primary extrauterine endometrial stromal sarcoma. Hum Pathol. 2011;42(7):939\u201346.","journal-title":"Hum Pathol"},{"issue":"3","key":"807_CR26","doi-asserted-by":"publisher","first-page":"349","DOI":"10.1007\/s00428-006-0345-8","volume":"450","author":"K Sato","year":"2007","unstructured":"Sato K, Ueda Y, Sugaya J, et al. Extrauterine endometrial stromal sarcoma with JAZF1\/JJAZ1 fusion confirmed by RT-PCR and interphase FISH presenting as an inguinal tumor. Virchows Arch. 2007;450(3):349\u201353.","journal-title":"Virchows Arch"},{"issue":"3","key":"807_CR27","doi-asserted-by":"publisher","first-page":"388","DOI":"10.1016\/S1525-1578(10)60568-5","volume":"7","author":"A Hrzenjak","year":"2005","unstructured":"Hrzenjak A, Moinfar F, Tavassoli FA, et al. JAZF1\/JJAZ1 gene fusion in endometrial stromal sarcomas: molecular analysis by reverse transcriptase-polymerase chain reaction optimized for paraffin-embedded tissue. J Mol Diagn. 2005;7(3):388\u201395.","journal-title":"J Mol Diagn"},{"issue":"2","key":"807_CR28","doi-asserted-by":"publisher","first-page":"183","DOI":"10.1002\/gcc.22132","volume":"53","author":"CR Antonescu","year":"2014","unstructured":"Antonescu CR, Sung YS, Chen CL, et al. Novel ZC3H7B-BCOR, MEAF6-PHF1, and EPC1-PHF1 fusions in ossifying fibromyxoid tumors--molecular characterization shows genetic overlap with endometrial stromal sarcoma. Genes Chromosom Cancer. 2014;53(2):183\u201393.","journal-title":"Genes Chromosom Cancer"},{"issue":"7","key":"807_CR29","doi-asserted-by":"publisher","first-page":"996","DOI":"10.1097\/PAS.0b013e318160736a","volume":"32","author":"M Miettinen","year":"2008","unstructured":"Miettinen M, Finnell V, Fetsch JF. Ossifying fibromyxoid tumor of soft parts--a clinicopathologic and immunohistochemical study of 104 cases with long-term follow-up and a critical review of the literature. Am J Surg Pathol. 2008;32(7):996\u20131005.","journal-title":"Am J Surg Pathol"},{"issue":"4","key":"807_CR30","doi-asserted-by":"publisher","first-page":"371","DOI":"10.5858\/arpa.2014-0590-RS","volume":"140","author":"D Bakiratharajan","year":"2016","unstructured":"Bakiratharajan D, Rekhi B. Ossifying Fibromyxoid tumor: an update. Arch Pathol Lab Med. 2016;140(4):371\u20135.","journal-title":"Arch Pathol Lab Med"},{"issue":"3","key":"807_CR31","doi-asserted-by":"publisher","first-page":"1069","DOI":"10.1016\/j.ajpath.2012.05.030","volume":"181","author":"S Gebre-Medhin","year":"2012","unstructured":"Gebre-Medhin S, Nord KH, M\u00f6ller E, et al. Recurrent rearrangement of the PHF1 gene in ossifying fibromyxoid tumors. Am J Pathol. 2012;181(3):1069\u201377.","journal-title":"Am J Pathol"}],"container-title":["Diagnostic Pathology"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1186\/s13000-019-0807-3.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/article\/10.1186\/s13000-019-0807-3\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1186\/s13000-019-0807-3.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2020,4,20]],"date-time":"2020-04-20T23:06:54Z","timestamp":1587424014000},"score":1,"resource":{"primary":{"URL":"https:\/\/diagnosticpathology.biomedcentral.com\/articles\/10.1186\/s13000-019-0807-3"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2019,4,22]]},"references-count":31,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2019,12]]}},"alternative-id":["807"],"URL":"https:\/\/doi.org\/10.1186\/s13000-019-0807-3","relation":{},"ISSN":["1746-1596"],"issn-type":[{"value":"1746-1596","type":"electronic"}],"subject":[],"published":{"date-parts":[[2019,4,22]]},"assertion":[{"value":"17 December 2018","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"4 April 2019","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"22 April 2019","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"University Health Network (UHN, Toronto, Canada) Research Ethics Board (REB) approval was granted (reference number: 16\u20135334.3).","order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"There is automatic institutional consent for all patients treated at our institution.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"Not applicable - The authors declare that they have no competing interests.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}},{"value":"Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Publisher\u2019s Note"}}],"article-number":"30"}}