{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,11]],"date-time":"2026-07-11T16:47:40Z","timestamp":1783788460757,"version":"3.55.0"},"reference-count":32,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2021,5,5]],"date-time":"2021-05-05T00:00:00Z","timestamp":1620172800000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2021,5,5]],"date-time":"2021-05-05T00:00:00Z","timestamp":1620172800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["J Med Case Reports"],"published-print":{"date-parts":[[2021,12]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec>\n                <jats:title>Background<\/jats:title>\n                <jats:p>Low-grade endometrial stromal sarcoma is a rare neoplastic growth in the uterine cavity, representing less than 1% of uterine tumors. Such tumors usually affect premenopausal and perimenopausal women, with a mean age of 46 years. Treatment generally starts with surgical resection of the tumor, followed by chemotherapy, radiotherapy, or hormonal therapy.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Case presentation<\/jats:title>\n                <jats:p>In the current report, we again present a case of low-grade endometrial stromal sarcoma in a 51-year-old Mediterranean woman presenting with abdominopelvic pain. Computed tomography scan revealed a primary uterine tumor measuring 17\u00a0\u00d7\u00a09\u00a0\u00d7\u00a09\u00a0cm metastasizing to the lungs, bladder, and ureteral orifice, along with lymphovascular involvement. The patient underwent total abdominal hysterectomy, omentectomy, and lymph node dissection. Estrogen deprivation was accomplished by bilateral salpingo-oophorectomy. Lifelong hormonal therapy consisting of letrozole 2.5\u00a0mg per day was prescribed, which demonstrated remarkable efficacy, resulting in a partial remission of lung metastasis within 8\u00a0months after surgery. Full remission was observed after 18\u00a0months of hormonal therapy, with no recurrence. Another scan was performed after 2.5\u00a0years, revealing complete remission with no recurrence.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Conclusion<\/jats:title>\n                <jats:p>We again report a case of complete remission of low-grade endometrial stromal sarcoma after surgical removal of the tumor along with first-line hormonal therapy without the use of chemotherapy or radiotherapy, emphasizing the role of hormonal therapy in the treatment of such tumors.<\/jats:p>\n              <\/jats:sec>","DOI":"10.1186\/s13256-021-02838-x","type":"journal-article","created":{"date-parts":[[2021,5,4]],"date-time":"2021-05-04T23:05:26Z","timestamp":1620169526000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":9,"title":["Complete remission of advanced low-grade endometrial stromal sarcoma after aromatase inhibitor therapy: a case report"],"prefix":"10.1186","volume":"15","author":[{"given":"Omar F.","family":"Altal","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ahmed H.","family":"Al Sharie","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Omar M.","family":"Halalsheh","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nour","family":"Tashtush","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sarat","family":"Shaban","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mahmoud","family":"Alfaqih","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Abdelwahab","family":"Aleshawi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2021,5,5]]},"reference":[{"issue":"1","key":"2838_CR1","doi-asserted-by":"publisher","first-page":"80","DOI":"10.1016\/j.ejogrb.2009.02.005","volume":"144","author":"T Dahhan","year":"2009","unstructured":"Dahhan T, Fons G, Buist MR, ten Kate FJW, van der Velden J. The efficacy of hormonal treatment for residual or recurrent low-grade endometrial stromal sarcoma. A retrospective study. Eur J Obstet Gynecol Reprod Biol. 2009;144(1):80\u20134.","journal-title":"Eur J Obstet Gynecol Reprod Biol"},{"issue":"1","key":"2838_CR2","doi-asserted-by":"publisher","first-page":"21","DOI":"10.4103\/0971-5851.68848","volume":"31","author":"G Puliyath","year":"2010","unstructured":"Puliyath G, Nair VR, Singh S. Endometrial stromal sarcoma. Indian J Med Paediatr Oncol. 2010;31(1):21\u20133.","journal-title":"Indian J Med Paediatr Oncol"},{"issue":"3","key":"2838_CR3","doi-asserted-by":"publisher","first-page":"676","DOI":"10.1097\/AOG.0b013e3182a189ac","volume":"122","author":"JA Rauh-Hain","year":"2013","unstructured":"Rauh-Hain JA, del Carmen MG. Endometrial stromal sarcoma: a systematic. Obstetr Gynecol. 2013;122(3):676\u201383.","journal-title":"Obstetr Gynecol"},{"issue":"2","key":"2838_CR4","doi-asserted-by":"publisher","first-page":"254","DOI":"10.1016\/j.ygyno.2017.05.036","volume":"146","author":"B-LL Seagle","year":"2017","unstructured":"Seagle B-LL, Shilpi A, Buchanan S, Goodman C, Shahabi S. Low-grade and high-grade endometrial stromal sarcoma: a national cancer database study. Gynecol Oncol. 2017;146(2):254\u201362.","journal-title":"Gynecol Oncol"},{"issue":"5","key":"2838_CR5","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":"2838_CR6","doi-asserted-by":"publisher","first-page":"95","DOI":"10.1293\/tox.2017-0056","volume":"31","author":"S Kumabe","year":"2018","unstructured":"Kumabe S, Sato J, Tomonari Y, Takahashi M, Inoue K, Yoshida M, Doi T, Wako Y, Tsuchitani M. Morphological and immunohistochemical diversity of endometrial stromal sarcoma in rats. J Toxicol Pathol. 2018;31(2):95\u2013103.","journal-title":"J Toxicol Pathol"},{"issue":"6","key":"2838_CR7","doi-asserted-by":"publisher","first-page":"383","DOI":"10.1097\/PAP.0000000000000046","volume":"21","author":"CMJ Conklin","year":"2014","unstructured":"Conklin CMJ, Longacre TA. Endometrial stromal tumors: the new WHO classification. Adv Anat Pathol. 2014;21(6):383\u201393.","journal-title":"Adv Anat Pathol"},{"issue":"1","key":"2838_CR8","doi-asserted-by":"publisher","first-page":"1","DOI":"10.4103\/0971-5851.96960","volume":"33","author":"G Puliyath","year":"2012","unstructured":"Puliyath G, Nair MK. Endometrial stromal sarcoma: a review of the literature. Indian J Med Paediatr Oncol. 2012;33(1):1\u20136.","journal-title":"Indian J Med Paediatr Oncol"},{"key":"2838_CR9","doi-asserted-by":"publisher","first-page":"218","DOI":"10.1016\/j.jclinepi.2017.04.026","volume":"89","author":"DS Riley","year":"2017","unstructured":"Riley DS, Barber MS, Kienle GS, Aronson JK, von Schoen-Angerer T, Tugwell P, Kiene H, Helfand M, Altman DG, Sox H, et al. CARE guidelines for case reports: explanation and elaboration document. J Clin Epidemiol. 2017;89:218\u201335.","journal-title":"J Clin Epidemiol"},{"issue":"1","key":"2838_CR10","doi-asserted-by":"publisher","first-page":"170","DOI":"10.1016\/S0090-8258(03)00258-0","volume":"90","author":"MC Chu","year":"2003","unstructured":"Chu MC, Mor G, Lim C, Zheng W, Parkash V, Schwartz PE. Low-grade endometrial stromal sarcoma: hormonal aspects. Gynecol Oncol. 2003;90(1):170\u20136.","journal-title":"Gynecol Oncol"},{"issue":"4","key":"2838_CR11","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"},{"issue":"2\u20134","key":"2838_CR12","doi-asserted-by":"publisher","first-page":"190","DOI":"10.1159\/000108300","volume":"118","author":"F Micci","year":"2007","unstructured":"Micci F, Heim S. Pathogenetic mechanisms in endometrial stromal sarcoma. Cytogenet Genome Res. 2007;118(2\u20134):190\u20135.","journal-title":"Cytogenet Genome Res"},{"issue":"2","key":"2838_CR13","doi-asserted-by":"publisher","first-page":"273","DOI":"10.1016\/0090-8258(90)90054-O","volume":"38","author":"MM Altaras","year":"1990","unstructured":"Altaras MM, Jaffe R, Cohen I, Gruber A, Yanai-Inbar I, Bernheim J. Role of prolonged excessive estrogen stimulation in the pathogenesis of endometrial sarcomas: two cases and a review of the literature. Gynecol Oncol. 1990;38(2):273\u20137.","journal-title":"Gynecol Oncol"},{"issue":"4","key":"2838_CR14","doi-asserted-by":"publisher","first-page":"1495","DOI":"10.3892\/ol.2015.2962","volume":"9","author":"R Hu","year":"2015","unstructured":"Hu R, Hilakivi-Clarke L, Clarke R. Molecular mechanisms of tamoxifen-associated endometrial cancer (Review). Oncol Lett. 2015;9(4):1495\u2013501.","journal-title":"Oncol Lett"},{"issue":"2","key":"2838_CR15","doi-asserted-by":"publisher","first-page":"297","DOI":"10.1016\/j.ygyno.2018.03.008","volume":"149","author":"LE Stewart","year":"2018","unstructured":"Stewart LE, Beck TL, Giannakopoulos NV, Rendi MH, Isacson C, Goff BA. Impact of oophorectomy and hormone suppression in low grade endometrial stromal sarcoma: a multicenter review. Gynecol Oncol. 2018;149(2):297\u2013300.","journal-title":"Gynecol Oncol"},{"issue":"4","key":"2838_CR16","doi-asserted-by":"publisher","first-page":"1189","DOI":"10.1002\/1097-0142(197904)43:4<1189::AID-CNCR2820430405>3.0.CO;2-E","volume":"43","author":"LM Lehrner","year":"1979","unstructured":"Lehrner LM, Miles PA, Enck RE. Complete remission of widely metastatic endometrial stromal sarcoma following combination chemotherapy. Cancer. 1979;43(4):1189\u201394.","journal-title":"Cancer"},{"issue":"7","key":"2838_CR17","doi-asserted-by":"publisher","first-page":"1232","DOI":"10.1111\/IGC.0b013e3181b33c9a","volume":"19","author":"B Barney","year":"2009","unstructured":"Barney B, Tward JD, Skidmore T, Gaffney DK. Does radiotherapy or lymphadenectomy improve survival in endometrial stromal sarcoma? Int J Gynecol Cancer. 2009;19(7):1232\u20138.","journal-title":"Int J Gynecol Cancer"},{"issue":"3\u20138","key":"2838_CR18","doi-asserted-by":"publisher","first-page":"589","DOI":"10.1016\/0960-0760(92)90389-Z","volume":"41","author":"D Navarro","year":"1992","unstructured":"Navarro D, Cabrera JJ, Le\u00f3n L, Chirino R, Fern\u00e1ndez L, L\u00f3pez A, Rivero JF, Fern\u00e1ndez P, Falc\u00f3n O, Jim\u00e9nez P, et al. Endometrial stromal sarcoma expression of estrogen receptors, progesterone receptors and estrogen-induced srp27 (24K) suggests hormone responsiveness. J Steroid Biochem Mol Biol. 1992;41(3\u20138):589\u201396.","journal-title":"J Steroid Biochem Mol Biol"},{"issue":"4","key":"2838_CR19","doi-asserted-by":"publisher","first-page":"347","DOI":"10.1097\/CCO.0b013e3281a7ef3a","volume":"19","author":"O Reich","year":"2007","unstructured":"Reich O, Regauer S. Hormonal therapy of endometrial stromal sarcoma. Curr Opin Oncol. 2007;19(4):347\u201352.","journal-title":"Curr Opin Oncol"},{"issue":"1","key":"2838_CR20","doi-asserted-by":"publisher","first-page":"8","DOI":"10.1186\/s13569-015-0024-0","volume":"5","author":"E Thanopoulou","year":"2015","unstructured":"Thanopoulou E, Aleksic A, Thway K, Khabra K, Judson I. Hormonal treatments in metastatic endometrial stromal sarcomas: the 10-year experience of the sarcoma unit of Royal Marsden Hospital. Clin Sarcoma Res. 2015;5(1):8.","journal-title":"Clin Sarcoma Res"},{"issue":"1","key":"2838_CR21","doi-asserted-by":"publisher","first-page":"87","DOI":"10.1385\/MO:20:1:87","volume":"20","author":"JP Spano","year":"2003","unstructured":"Spano JP, Soria JC, Kambouchner M, Piperno-Neuman S, Morin F, Morere JF, Martin A, Breau JL. Long-term survival of patients given hormonal therapy for metastatic endometrial stromal sarcoma. Med Oncol. 2003;20(1):87.","journal-title":"Med Oncol"},{"issue":"5","key":"2838_CR22","doi-asserted-by":"publisher","first-page":"600","DOI":"10.1177\/1933719118778801","volume":"26","author":"U Deshmukh","year":"2019","unstructured":"Deshmukh U, Black J, Perez-Irizarry J, Passarelli R, Levy K, Rostkowski A, Hui P, Rutherford TJ, Santin AD, Azodi M, et al. Adjuvant hormonal therapy for low-grade endometrial stromal sarcoma. Reprod Sci. 2019;26(5):600\u20138.","journal-title":"Reprod Sci"},{"issue":"4","key":"2838_CR23","doi-asserted-by":"publisher","first-page":"275","DOI":"10.1097\/00001813-200004000-00008","volume":"11","author":"PW Szlosarek","year":"2000","unstructured":"Szlosarek PW, Lofts FJ, Pettengell R, Carter P, Young M, Harmer C. Effective treatment of a patient with a high-grade endometrial stromal sarcoma with an accelerated regimen of carboplatin and paclitaxel. Anticancer Drugs. 2000;11(4):275\u20138.","journal-title":"Anticancer Drugs"},{"issue":"5","key":"2838_CR24","doi-asserted-by":"publisher","first-page":"897","DOI":"10.1007\/s10147-013-0627-5","volume":"19","author":"T Takano","year":"2014","unstructured":"Takano T, Niikura H, Ito K, Nagase S, Utsunomiya H, Otsuki T, Toyoshima M, Tokunaga H, Kaiho-Sakuma M, Shiga N, et al. Feasibility study of gemcitabine plus docetaxel in advanced or recurrent uterine leiomyosarcoma and undifferentiated endometrial sarcoma in Japan. Int J Clin Oncol. 2014;19(5):897\u2013905.","journal-title":"Int J Clin Oncol"},{"issue":"1","key":"2838_CR25","doi-asserted-by":"publisher","first-page":"e3","DOI":"10.3802\/jgo.2018.29.e3","volume":"29","author":"HJ Kim","year":"2018","unstructured":"Kim HJ, Kim Y, Lee SJ, Lee J, Park SH. Pazopanib monotherapy in the treatment of pretreated, metastatic uterine sarcoma: a single-center retrospective study. J Gynecol Oncol. 2018;29(1):e3.","journal-title":"J Gynecol Oncol"},{"issue":"6","key":"2838_CR26","first-page":"2327","volume":"24","author":"V Sunar","year":"2019","unstructured":"Sunar V, Korkmaz V, Akin S, Can Guven D, Arik Z, Ates O, Yilmaz M, Mutlu Meydanli M, Oksuzoglu B. Efficacy of Pazopanib in patients with metastatic uterine sarcoma: a multi-institutional study. J BUON. 2019;24(6):2327\u201332.","journal-title":"J BUON"},{"issue":"1","key":"2838_CR27","doi-asserted-by":"publisher","first-page":"92","DOI":"10.1186\/s12885-018-3999-0","volume":"18","author":"AJ Verschoor","year":"2018","unstructured":"Verschoor AJ, Warmerdam FARM, Bosse T, Bov\u00e9e JVMG, Gelderblom H. A remarkable response to pazopanib, despite recurrent liver toxicity, in a patient with a high grade endometrial stromal sarcoma, a case report. BMC Cancer. 2018;18(1):92.","journal-title":"BMC Cancer"},{"issue":"1","key":"2838_CR28","doi-asserted-by":"publisher","first-page":"57","DOI":"10.1007\/s00404-008-0631-6","volume":"279","author":"\u00d6 Alkasi","year":"2009","unstructured":"Alkasi \u00d6, Meinhold-Heerlein I, Zaki R, Fasching P, Maass N, Jonat W, Beckmann MW. Long-term disease-free survival after hormonal therapy of a patient with recurrent low grade endometrial stromal sarcoma: a case report. Arch Gynecol Obstet. 2009;279(1):57\u201360.","journal-title":"Arch Gynecol Obstet"},{"issue":"3","key":"2838_CR29","doi-asserted-by":"publisher","first-page":"769","DOI":"10.1016\/j.ygyno.2004.07.063","volume":"95","author":"M Leunen","year":"2004","unstructured":"Leunen M, Breugelmans M, De Sutter P, Bourgain C, Amy JJ. Low-grade endometrial stromal sarcoma treated with the aromatase inhibitor letrozole. Gynecol Oncol. 2004;95(3):769\u201371.","journal-title":"Gynecol Oncol"},{"issue":"3","key":"2838_CR30","doi-asserted-by":"publisher","first-page":"771","DOI":"10.1007\/s12032-010-9511-6","volume":"28","author":"K Shoji","year":"2011","unstructured":"Shoji K, Oda K, Nakagawa S, Kawana K, Yasugi T, Ikeda Y, Takazawa Y, Kozuma S, Taketani Y. Aromatase inhibitor anastrozole as a second-line hormonal treatment to a recurrent low-grade endometrial stromal sarcoma: a case report. Med Oncol. 2011;28(3):771\u20134.","journal-title":"Med Oncol"},{"issue":"3","key":"2838_CR31","doi-asserted-by":"publisher","first-page":"458","DOI":"10.1006\/gyno.1998.5174","volume":"71","author":"DR Scribner","year":"1998","unstructured":"Scribner DR, Walker JL. Low-grade endometrial stromal sarcoma preoperative treatment with Depo-Lupron and Megace. Gynecol Oncol. 1998;71(3):458\u201360.","journal-title":"Gynecol Oncol"},{"issue":"7","key":"2838_CR32","first-page":"e9249","volume":"12","author":"M Gangireddy","year":"2020","unstructured":"Gangireddy M, Chan Gomez J, Kanderi T, Joseph M, Kundoor V. Recurrence of endometrial stromal sarcoma, two decades post-treatment. Cureus. 2020;12(7):e9249.","journal-title":"Cureus"}],"container-title":["Journal of Medical Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s13256-021-02838-x.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s13256-021-02838-x\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s13256-021-02838-x.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,5,4]],"date-time":"2021-05-04T23:19:30Z","timestamp":1620170370000},"score":1,"resource":{"primary":{"URL":"https:\/\/jmedicalcasereports.biomedcentral.com\/articles\/10.1186\/s13256-021-02838-x"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,5,5]]},"references-count":32,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2021,12]]}},"alternative-id":["2838"],"URL":"https:\/\/doi.org\/10.1186\/s13256-021-02838-x","relation":{},"ISSN":["1752-1947"],"issn-type":[{"value":"1752-1947","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,5,5]]},"assertion":[{"value":"20 March 2020","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"29 March 2021","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"5 May 2021","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"Ethical approval for case reports is waived by the institutional review board (IRB) in the King Abdullah University Hospital (KAUH). Consent to participate is not applicable.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare that they have no competing interests.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"262"}}