{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,8]],"date-time":"2025-10-08T04:40:50Z","timestamp":1759898450365,"version":"build-2065373602"},"reference-count":46,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2025,10,8]],"date-time":"2025-10-08T00:00:00Z","timestamp":1759881600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Oncol."],"abstract":"<jats:sec><jats:title>Introduction<\/jats:title><jats:p>Low-grade endometrial stromal sarcoma (LGESS) is a rare malignant tumor of the uterus, characterized by slow growth. Early-stage LGESS is associated with favorable survival, but it has a high recurrence rate. The primary treatment for this disease is full-staging surgery. In this report, we present a case to explore the potential for fertility-preserving treatment in young women with LGESS.<\/jats:p><\/jats:sec><jats:sec><jats:title>Case presentation<\/jats:title><jats:p>A 29-year-old nulliparous patient diagnosed with stage IA LGESS underwent conservative treatment at the Obstetrics and Gynecology Hospital of Fudan University. After fertility-sparing surgery and three months of hormone treatment, no residual lesions were found. Subsequently, the patient conceived spontaneously and successfully delivered a healthy baby. However, she experienced recurrence eight months after delivery but declined hysterectomy and follow-up care.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>Currently, there is still no standard management protocol for fertility preservation therapy in LGESS. Both previously reported cases and our case suggest that fertility-sparing treatment may be an option for carefully selected patients with LGESS. Further research and larger clinical studies are necessary to explore fertility-preserving treatments for young nulliparous patients with LGESS to establish guidelines or consensus.<\/jats:p><\/jats:sec>","DOI":"10.3389\/fonc.2025.1572914","type":"journal-article","created":{"date-parts":[[2025,10,8]],"date-time":"2025-10-08T04:18:00Z","timestamp":1759897080000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":0,"title":["Case Report: Spontaneous pregnancy after fertility-preserving treatment in a patient with low-grade endometrial stromal sarcoma and literature review"],"prefix":"10.3389","volume":"15","author":[{"given":"Manrong","family":"Wang","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Pengfei","family":"Wu","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lulu","family":"Wang","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sijia","family":"Liu","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Qujia","family":"Gama","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Qiaoying","family":"Lv","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jinyu","family":"Zhang","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Min","family":"Yu","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yiqin","family":"Wang","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Fenghua","family":"Ma","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Weiwei","family":"Shan","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Xuezhen","family":"Luo","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1965","published-online":{"date-parts":[[2025,10,8]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","DOI":"10.3760\/cma.j.issn.0529-5807.2014.10.001","article-title":"Introduction of WHO classification of tumours of female reproductive organs, fourth edition","volume":"43","author":"Lu","year":"2014","journal-title":"Zhonghua Bing Li Xue Za Zhi"},{"key":"B2","doi-asserted-by":"publisher","DOI":"10.1097\/PAP.0000000000000046","article-title":"Endometrial stromal tumors: The new WHO classification","volume":"21","author":"Conklin","year":"2014","journal-title":"Adv Anat Pathol"},{"key":"B3","doi-asserted-by":"publisher","DOI":"10.1111\/his.14609","article-title":"Key changes to the world health organization (WHO) classification of female genital tumours introduced in the 5th edition (2020)","volume":"80","author":"McCluggage","year":"2022","journal-title":"Histopathology"},{"key":"B4","article-title":"National Comprehensive Cancer Network (NCCN). NCCN guidelines version 2","author":"Abu-Rustum","year":"2024","journal-title":"Uterine Neoplasms. [Internet]"},{"key":"B5","doi-asserted-by":"publisher","first-page":"95","DOI":"10.1016\/j.ygyno.2023.02.009","article-title":"Endometrial stromal tumors of the uterus: Epidemiology, pathological and biological features, treatment options and clinical outcomes","volume":"171","author":"Gadducci","year":"2023","journal-title":"Gynecol Oncol"},{"key":"B6","doi-asserted-by":"publisher","DOI":"10.1016\/j.gore.2024.101470","article-title":"Uterine preservation in low-grade endometrial stromal sarcoma","volume":"55","author":"Rajaram","year":"2024","journal-title":"Gynecol Oncol Rep"},{"key":"B7","doi-asserted-by":"publisher","DOI":"10.1136\/ijgc-2023\u2013004448","article-title":"Fertility-sparing management of low-grade endometrial stromal sarcoma","volume":"33","author":"Laufer","year":"2023","journal-title":"Int J Gynecol Cancer"},{"key":"B8","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijscr.2023.108857","article-title":"A case of a recurrent low-grade endometrial stromal sarcoma extending to the inferior vena cava (IVC) after the primary fertility-sparing surgery","volume":"111","author":"Yano","year":"2023","journal-title":"Int J Surg Case Rep"},{"key":"B9","doi-asserted-by":"publisher","DOI":"10.3390\/jcm11164761","article-title":"Fertility-sparing management may be considered in young women with uterine sarcoma","volume":"11","author":"Pi\u0105tek","year":"2022","journal-title":"J Clin Med"},{"key":"B10","doi-asserted-by":"publisher","DOI":"10.3389\/fonc.2022.922757","article-title":"Hormone therapy reduces recurrence in stage II-IV uterine low-grade endometrial stromal sarcomas: A retrospective cohort study","volume":"12","author":"Huang","year":"2022","journal-title":"Front Oncol"},{"key":"B11","doi-asserted-by":"publisher","DOI":"10.12998\/wjcc.v9.i4.983","article-title":"Fertility-sparing surgeries without adjuvant therapy through term pregnancies in a patient with low-grade endometrial stromal sarcoma: A case report","volume":"9","author":"Gu","year":"2021","journal-title":"World J Clin Cases"},{"key":"B12","doi-asserted-by":"publisher","DOI":"10.18632\/oncotarget.12491","article-title":"Fertility-sparing surgery for patients with low-grade endometrial stromal sarcoma","volume":"8","author":"Xie","year":"2017","journal-title":"Oncotarget"},{"key":"B13","doi-asserted-by":"publisher","first-page":"1358","DOI":"10.21037\/atm-20\u20132180","article-title":"Fertility-sparing management of low-grade endometrial stromal sarcoma: Analysis of an institutional series, a population-based analysis and review of the literature","volume":"8","author":"Zheng","year":"2020","journal-title":"Ann Transl Med"},{"key":"B14","doi-asserted-by":"publisher","DOI":"10.1007\/s11655-020-3251-z","article-title":"Natural pregnancy and successful delivery in a woman with endometrial stromal sarcoma after sequential therapy with heavy dosage of Chinese medicine: A case report","volume":"26","author":"Xie","year":"2020","journal-title":"Chin J Integr Med"},{"key":"B15","doi-asserted-by":"publisher","DOI":"10.1111\/jog.13695","article-title":"Recurrence after fertility-preserving surgery for low-grade endometrial stromal sarcoma","volume":"44","author":"Chin","year":"2018","journal-title":"J Obstet Gynaecol Res"},{"key":"B16","doi-asserted-by":"publisher","first-page":"233","DOI":"10.1186\/s13256-015-0719\u20130","article-title":"Long-term survival in low-grade endometrial stromal sarcoma with childbirth and multidisciplinary treatment: A case report","volume":"9","author":"Maeda","year":"2015","journal-title":"J Med Case Rep"},{"key":"B17","doi-asserted-by":"publisher","first-page":"29","DOI":"10.2147\/OTT.S69507","article-title":"Fertility sparing surgery in young women affected by endometrial stromal sarcoma: An oncologic dilemma or a reliable option? review of literature starting from a peculiar case","volume":"8","author":"Noventa","year":"2015","journal-title":"Onco Targets Ther"},{"key":"B18","doi-asserted-by":"publisher","DOI":"10.1111\/jog.12506","article-title":"Fatal case of multiple recurrences of endometrial stromal sarcoma after fertility-sparing management","volume":"41","author":"Morimoto","year":"2015","journal-title":"J Obstet Gynaecol Res"},{"key":"B19","article-title":"Fertility-sparing treatment of low-grade endometrial stromal sarcoma","volume":"8","author":"Jin","year":"2015","journal-title":"Int J Clin Exp Med"},{"key":"B20","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejogrb.2015.09.041","article-title":"Fertility-sparing management of low-grade endometrial stromal sarcoma: Analysis of an institutional series and review of the literature","volume":"195","author":"Laurelli","year":"2015","journal-title":"Eur J Obstet Gynecol Reprod Biol"},{"key":"B21","doi-asserted-by":"publisher","DOI":"10.1007\/s13224-012-0249-2","article-title":"Successful pregnancy with endometrial stromal sarcoma (ESS)","volume":"64","author":"Jain","year":"2014","journal-title":"J Obstet Gynecol India"},{"key":"B22","doi-asserted-by":"publisher","DOI":"10.1016\/j.ygyno.2013.12.032","article-title":"Ovary and uterus-sparing procedures for low-grade endometrial stromal sarcoma: A retrospective study of 153 cases","volume":"132","author":"Bai","year":"2014","journal-title":"Gynecol Oncol"},{"key":"B23","doi-asserted-by":"publisher","DOI":"10.1016\/j.pdpdt.2014.07.007","article-title":"Fertility-sparing management combined with photodynamic therapy for endometrial stromal sarcoma: A case report","volume":"11","author":"Choi","year":"2014","journal-title":"Photodiagnosis Photodyn Ther"},{"key":"B24","doi-asserted-by":"publisher","DOI":"10.3760\/cma.j.issn.0366-6999.20131530","article-title":"Successful term pregnancy after laparoscopic surgery of low grade endometrial stromal sarcoma","volume":"127","author":"Zhan","year":"2014","journal-title":"Chin Med J (Engl)"},{"key":"B25","doi-asserted-by":"publisher","DOI":"10.3892\/ol.2014.2288","article-title":"Conservative management of endometrial stromal sarcoma at stage III: A case report","volume":"8","author":"Dong","year":"2014","journal-title":"Oncol Lett"},{"key":"B26","doi-asserted-by":"publisher","DOI":"10.1097\/AOG.0b013e31825a7397","article-title":"Successful pregnancy after fertility-sparing local resection and uterine reconstruction for low-grade endometrial stromal sarcoma","volume":"120","author":"Delaney","year":"2012","journal-title":"Obstet Gynecol"},{"key":"B27","doi-asserted-by":"publisher","first-page":"4","DOI":"10.1016\/j.gynor.2012.10.001","article-title":"Reproductive outcome after uterine-sparing surgery for endometrial stromal sarcoma","volume":"3","author":"S\u00e1nchez-Ferrer","year":"2012","journal-title":"Gynecol Oncol Case Rep"},{"key":"B28","article-title":"Conservative management of low-grade endometrial stromal sarcoma followed by pregnancy and severe recurrence","volume":"29","author":"Koskas","year":"2009","journal-title":"Anticancer Res"},{"key":"B29","doi-asserted-by":"publisher","DOI":"10.1016\/j.ygyno.2005.06.039","article-title":"Conservative management of a myxoid endometrial stromal sarcoma in a 16-year-old nulliparous woman","volume":"99","author":"Stadsvold","year":"2005","journal-title":"Gynecol Oncol"},{"key":"B30","doi-asserted-by":"publisher","DOI":"10.1046\/j.1525-1438.1997.00012.x","article-title":"Conservative management of endometrial stromalsarcoma in young women","volume":"7","author":"Lissoni","year":"2010","journal-title":"Int J Gynecological Cancer"},{"key":"B31","doi-asserted-by":"publisher","DOI":"10.1016\/0090-8258(90)90120-a","article-title":"Endometrial stromal sarcomas","volume":"36","author":"Mansi","year":"1990","journal-title":"Gynecol Oncol"},{"key":"B32","doi-asserted-by":"publisher","DOI":"10.1007\/s003300051003","article-title":"Endometrial stromal sarcoma of the uterus: MR and US findings","volume":"10","author":"Gandolfo","year":"2000","journal-title":"Eur Radiol"},{"key":"B33","doi-asserted-by":"publisher","DOI":"10.3390\/nu11122885","article-title":"Threshold-effect association of dietary cholesterol intake with dyslipidemia in Chinese adults: Results from the China health and nutrition survey in 2015","volume":"11","author":"Huang","year":"2019","journal-title":"Nutrients"},{"key":"B34","doi-asserted-by":"publisher","DOI":"10.1016\/j.bpobgyn.2011.07.004","article-title":"Endometrial stromal sarcoma of uterus","volume":"25","author":"Xue","year":"2011","journal-title":"Best Pract Res Clin Obstet Gynaecol"},{"key":"B35","doi-asserted-by":"publisher","DOI":"10.1016\/j.ygyno.2009.08.014","article-title":"Hormone receptor expression in uterine sarcomas: Prognostic and therapeutic roles","volume":"115","author":"Ioffe","year":"2009","journal-title":"Gynecol Oncol"},{"key":"B36","doi-asserted-by":"publisher","DOI":"10.1097\/PAS.0b013e3182262743","article-title":"Frequency of known gene rearrangements in endometrial stromal tumors","volume":"35","author":"Chiang","year":"2011","journal-title":"Am J Surg Pathol"},{"key":"B37","doi-asserted-by":"publisher","DOI":"10.1097\/PAS.0b013e318272c612","article-title":"Endometrial stromal sarcomas with sex cord differentiation are associated with PHF1 rearrangement","volume":"37","author":"D\u2019Angelo","year":"2013","journal-title":"Am J Surg Pathol"},{"key":"B38","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.101132598","article-title":"Frequent fusion of the JAZF1 and JJAZ1 genes in endometrial stromal tumors","volume":"98","author":"Koontz","year":"2001","journal-title":"Proc Natl Acad Sci USA"},{"key":"B39","doi-asserted-by":"publisher","DOI":"10.1016\/j.canlet.2014.01.030","article-title":"MEAF6\/PHF1 is a recurrent gene fusion in endometrial stromal sarcoma","volume":"347","author":"Micci","year":"2014","journal-title":"Cancer Lett"},{"key":"B40","doi-asserted-by":"publisher","DOI":"10.1002\/gcc.22907","article-title":"Molecular pathogenesis and prognostication of \u201clow-grade\u2019\u2019 and \u201chigh-grade\u201d endometrial stromal sarcoma","volume":"60","author":"Micci","year":"2021","journal-title":"Genes Chromosomes Cancer"},{"key":"B41","doi-asserted-by":"publisher","DOI":"10.1016\/j.labinv.2025.104198","article-title":"Decoding the molecular landscape of 262 uterine sarcomas: RNA-seq clustering of endometrial stromal sarcomas, uterine tumors resembling ovarian sex cord tumors, and undifferentiated uterine sarcomas with prognostic insights","volume":"105","author":"Hojn\u00fd","year":"2025","journal-title":"Lab Invest"},{"key":"B42","doi-asserted-by":"publisher","DOI":"10.3389\/fonc.2022.883344","article-title":"Low-grade uterine endometrial stromal sarcoma: prognostic analysis of clinico-pathological characteristics, surgical management, and adjuvant treatments. Experience From Two Referral Centers","volume":"12","author":"Borella","year":"2022","journal-title":"Front Oncol"},{"key":"B43","doi-asserted-by":"publisher","first-page":"12","DOI":"10.1136\/ijgc-2020-002230","article-title":"ESGO\/ESTRO\/ESP guidelines for the management of patients with endometrial carcinoma","volume":"31","author":"Concin","year":"2021","journal-title":"Int J Gynecol Cancer"},{"key":"B44","doi-asserted-by":"publisher","DOI":"10.1038\/sj.bjc.6603986","article-title":"Clinical study investigating the role of lymphadenectomy, surgical castration and adjuvant hormonal treatment in endometrial stromal sarcoma","volume":"97","author":"Amant","year":"2007","journal-title":"Br J Cancer"},{"key":"B45","doi-asserted-by":"publisher","DOI":"10.3892\/ol.2016.5186","article-title":"Letrozole as second-line hormonal treatment for recurrent low-grade endometrial stromal sarcoma: A case report and review of the literature","volume":"12","author":"Nakamura","year":"2016","journal-title":"Oncol Lett"},{"key":"B46","doi-asserted-by":"publisher","DOI":"10.1007\/s12672-010-0007\u20139","article-title":"Treatment of recurrent endometrial stromal sarcoma with letrozole: A case report and literature review","volume":"1","author":"Sylvestre","year":"2010","journal-title":"Horm Cancer"}],"container-title":["Frontiers in Oncology"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fonc.2025.1572914\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,8]],"date-time":"2025-10-08T04:18:02Z","timestamp":1759897082000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fonc.2025.1572914\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,10,8]]},"references-count":46,"alternative-id":["10.3389\/fonc.2025.1572914"],"URL":"https:\/\/doi.org\/10.3389\/fonc.2025.1572914","relation":{},"ISSN":["2234-943X"],"issn-type":[{"value":"2234-943X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,10,8]]},"article-number":"1572914"}}