Uterine leiomyosarcoma

Author(s): Giorgio Boganiaa; Giuseppe Carusob,c,d,†; Isabelle Ray-Coquarde; Pedro T. Ramirezf; Nicole Concing,h; Natalie YL. Ngoii; Robert L. Colemanj; Andrea Marianic; William Clibyc; Mario M. Leitao, Jr.k; Nadeem Abu Rustumk,l; Paolo G. Casalia; Alessandro Gronchia; Ignace Vergotem; Judith Kroepn; Takayuki Enomotoo; Kazuhiro Takeharap; Hannelore Denysq; Masashi Takanor; Diane Provenchers; Pauline Wimbergert; Se Ik Kimu; Jae-Weon Kimu; Gian Franco Zannoniv; David SP. Tani; Jvan Casarinw; Biagio Paolinia; Valentina Chiappaa; Francesco Raspagliesia; Ilaria Cuccua; Violante Di Donatox; David M. O’Malleyy; David Mutchz; Jubilee Brownaa; Fernanda Herrerabb; Nicoletta Colombob,cc; Sandro Pignatadd; Giovanni Scambiaee; Brian M. Slomovitzff; Bradley J. Monkgg
Source: DOI: 10.1016/j.ijgc.2025.101992

Dr. Anjan Patel's Thoughts

Shout out to Dr. Monk who is the first author on this study! Uterine leiomyosarcoma (uLMS) is a rare, aggressive uterine malignancy with a 5-year OS ranging from 51–76% in FIGO stage I to just 10–15% in stage IV, and a median OS of 10 months for advanced disease. The mainstay of treatment for localized disease is en-bloc TH ± BSO, with observation recommended postoperatively for stage I; adjuvant chemo or RT has not shown a clear survival benefit in early-stage disease. For advanced or unresectable cases, doxorubicin-based regimens (± trabectedin) remain standard, but even with multimodal therapy, outcomes remain poor (5-year recurrence 40–75%, 70% distant). Novel strategies—anti-angiogenics, ICIs, PARP inhibitors—are under investigation, but robust data are lacking. Bottom line: uLMS continues to challenge us with its aggressive biology and limited therapeutic gains, so we need to keep pushing for trial enrollment and molecularly targeted approaches.

ABSTRACT

Uterine leiomyosarcoma is a rare and heterogeneous gynecological malignancy that poses a significant clinical challenge due to its aggressive nature and limited treatment options. Its multifactorial etiopathogenesis involves complex cytogenetic and molecular aberrations, including TP53, RB1, and chromothripsis-associated gene alterations. The non-specific clinical presentation, resembling other benign conditions, complicates early and accurate diagnosis, alongside intricate radiological and pathological patterns. Advanced imaging techniques, such as magnetic resonance imaging and computed tomography, are employed to differentiate uterine leiomyosarcoma from benign conditions, but no single test is definitive. For FIGO (International Federation of Gynecology and Obstetrics) stage I uterine leiomyosarcoma, treatment consists of en bloc total hysterectomy ± bilateral salpingo-oophorectomy. Patients with stage II to IV disease amenable to complete resection can undergo surgery followed by adjuvant systemic therapy and/or radiotherapy. Lymphadenectomy is unnecessary in patients lacking bulky nodes. Unresectable or unsuitable cases warrant primary systemic therapy and/or radiotherapy. Managing recurrent disease requires a multimodal approach tailored to factors such as the site and number of metastases, prior radiotherapy, and resectability. Multidisciplinary management and centralization in referral centers are crucial for individualized decision-making. Ongoing research explores the intricate cytogenetic and molecular aberrations of uterine leiomyosarcoma, paving the way for personalized treatment strategies. This review, developed following the European Society of Gynaecological Oncology/Gynecologic Cancer InterGroup/European Reference Network on Rare Adult Solid Cancers guidelines, explores the clinical presentation, diagnostic challenges, and evolving therapeutic strategies for uterine leiomyosarcoma, while also highlighting variations in clinical practice worldwide.

Author Affiliations

aFondazione IRCCS Istituto Nazionale dei Tumori di Milano, Department of Surgery, Milan, Italy; bEuropean Institute of Oncology IRCCS, IEO, Division of Gynecologic Oncology, Milan, Italy; cMayo Clinic, Department of Obstetrics and Gynecology, Division of Gynecologic Oncology, Rochester, MN, USA; dSapienza University of Rome, Department of Experimental Medicine, Rome, Italy; eUniversité Claude Bernard Lyon Est, EA 7425, Centre Leon Bérard, Hesper lab, Lyon, France; fHouston Methodist Hospital Neal Cancer Center, Department of Obstetrics and Gynaecology, Houston, TX, USA; gInnsbruck Medical University, Department of Gynecology and Obstetrics, Innsbruck, Austria; hEvangelische Kliniken Essen-Mitte, Department of Gynecology and Gynecological Oncology, Essen, Germany; iNational University Cancer Institute, Singapore, Singapore; jUS Oncology Network, Texas Oncology, The Woodlands, TX, USA; kMemorial Sloan Kettering Cancer Center, Department of Surgery, Gynecology Service, New York, NY, USA; lJoan & Sanford I. Weill Medical College of Cornell University, New York, NY, USA; mCatholic University Leuven, University Hospitals Leuven, Gynaecological Oncology, Leuven, Belgium; nLeiden University Medical Center, Department of Medical Oncology, Leiden, the Netherlands; oOsaka University Graduate School of Medicine, Department of Obstetrics and Gynecology, Osaka, Japan; pNHO Shikoku Cancer Center, Department of Gynecologic Oncology, Matsuyama, Japan; qGhent University Hospital, Department of Medical Oncology, Ghent, Belgium; rNational Defense Medical College, Department of Obstetrics and Gynecology, Saitama, Japan; sCentre de Recherche du Centre hospitalier de l’Université de Montréal, Montreal, QC, Canada; tTechnische Universität Dresden, Department of Gyncology and Obstetrics, NCT Dresden, Dresden, Germany; uSeoul National University College of Medicine, Department of Obstetrics and Gynecology, Seoul, Republic of Korea; vLargo Francesco Vito, Università Cattolica del Sacro Cuore, Policlinico Universitario “Agostino Gemelli”-IRCCS, Division of Anatomic Pathology and Histology-Fondazione, Rome, Italy; wUniversity of Insubria, ‘Filippo Del Ponte’ Hospital, Department of Obstetrics and Gynecology, Varese, Italy; xPoliclinico Umberto I, Sapienza University of Rome, Department of Maternal and Child Health and Urological Sciences, Rome, Italy; yThe Ohio State University, College of Medicine, Department of Obstetrics and Gynecology, Division of Gynecologic Oncology, Columbus, OH, USA; zWashington University in Saint Louis, St Louis, MO, USA; aaAtrium Health, Levine Cancer Institute, Charlotte, NC, USA; bbCentre Hospitalier Universitaire Vaudois, Departement doncologie CHUV-UNIL, Lausanne, Switzerland; ccUniversity of Milan-Bicocca, Department of Medicine and Surgery, Milan, Italy; ddIstituto Nazionale Tumori IRCCS Fondazione G. Pascale, Department of Urology and Gynecology, Napoli, Italy; eeFondazione Policlinico Universitario A Gemelli IRCCS and Catholic University of Sacred Heart, Scientific Directorate, Rome, Italy; ffMount Sinai Medical Center, GOG Foundation, Miami Beach, FL, USA; ggFlorida Cancer Specialists and Research Institute, GOG Foundation, West Palm Beach, FL, USA; Giuseppe Caruso is Co-First author.

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