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      Venous Thromboembolism in Metastatic Uterine Leiomyosarcoma: A Case Report and Review of the Literature

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          Abstract

          We report an unusual case of extensive deep vein thrombosis (DVT) and pulmonary embolism (PE) in the setting of metastatic uterine leiomyosarcoma. Recognition of the associated sequelae of this condition may improve short- and long-term outcomes. A 56-year-old black female with a history of uterine leiomyosarcoma diagnosed incidentally after total abdominal hysterectomy for fibroid uterus without initiation of chemoradiation treatment presented to the emergency department complaining of generalized weakness and progressively worsening stridor for 2 weeks. The patient was experiencing shortness of breath, dysphagia, and hoarseness. Physical exam was remarkable for rhonchi but was otherwise normal. Diagnostic imaging via CT of the abdomen, pelvis, and chest revealed DVTs of the left common and external iliac veins, the superior mesenteric artery, multiple pulmonary emboli of the right pulmonary artery, several nodular lesions within the lungs, and scattered peritoneal necrotic lesions, which were suspicious for metastatic disease. Additionally, CT of the neck showed an exophytic mass protruding into the airway from the subglottic region and thyromegaly with bilateral thyroid lobe nodules. The patient was subsequently started on Eliquis and chemotherapy. The rarity of this case is rooted in the extent of the patient’s DVTs and PEs secondary to hypercoagulability in metastatic cancer. This presentation should be further evaluated to exclude thrombophilias or underlying malignancies. Drawing from the lessons of this case will help guide future clinical management regarding the care of metastatic uterine leiomyosarcoma.

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          Most cited references20

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          Thromboembolism is a leading cause of death in cancer patients receiving outpatient chemotherapy.

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            Prognostic factors and survival in 1396 patients with uterine leiomyosarcomas: emphasis on impact of lymphadenectomy and oophorectomy.

            The objectives of the current study were to determine the prognostic factors associated with disease-specific survival (DSS) and to analyze the role of lymphadenectomy (LND) and oophorectomy in the management of uterine leiomyosarcomas (LMS). Data were abstracted from the Surveillance, Epidemiology, and End Results database (1988-2003). Kaplan-Meier and Cox proportional hazards regression models were used for analyses. The median age of the 1396 patients was 52 years. There were 951 patients (68.1%) with International Federation of Gynecology and Obstetrics (FIGO) stage I disease, 43 patients (3.1%) with stage II disease, 99 patients (7.1%) with stage III disease, and 303 patients (21.7%) with stage IV disease. Distribution by tumor grade included 87 patients with grade 1 tumors, 208 with grade 2, and 509 patients with grade 3 tumors. The 5-year DSS rates for patients with stage I, II, III, and IV disease were 75.8%, 60.1%, 44.9%, and 28.7%, respectively. Lymph node metastases were identified in 23 of 348 patients (6.6%) who underwent LND. The 5-year DSS rate was 26% in patients who had positive lymph nodes compared with 64.2% in patients who had negative lymph nodes (P < .001). Of 341 patients aged <50 years with stage I or II disease, 240 (70.4%) underwent oophorectomy. There was no difference in 5-year DSS based on oophorectomy. On multivariate analysis, older age at diagnosis, more recent year of diagnosis, African-American race, higher tumor grade, higher stage of disease, and lack of primary surgical treatment all were associated significantly with worse survival. Independent predictors of DSS in patients with uterine LMS included age, race, stage, grade, and primary surgery. Oophorectomy was not found to have an independent impact on survival. Cancer 2008. (c) 2008 American Cancer Society.
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              Fixed-dose rate gemcitabine plus docetaxel as second-line therapy for metastatic uterine leiomyosarcoma: a Gynecologic Oncology Group phase II study.

              Doxorubicin-based treatment is standard therapy for metastatic uterine leiomyosarcoma. There is no standard second-line therapy. We determined activity of fixed-dose rate gemcitabine plus docetaxel as second-line treatment for metastatic uterine leiomyosarcoma. Eligible women with unresectable uterine leiomyosarcoma progressing after prior cytotoxic therapy were treated with gemcitabine 900 mg/m(2) days one and eight over 90 min, plus docetaxel 100 mg/m(2) on day 8 of a 21-day cycle with granulocyte growth factor. Patients with prior pelvic radiation received lower doses. Response Evaluation Criteria in Solid Tumors (RECIST) response was assessed by computed tomography (CT). Forty-eight of 51 women were evaluable for response (one wrong histology, two never treated). Prior therapy was doxorubicin-based in 90%, and ifosfamide-based in 6%. The overall objective response rate is 27%, with complete response in 6.3% (3/48), and partial response in 20.8% (10/48). An additional 50% (24/48) had stable disease (median duration 5.4 months). The median number of cycles per patient was 5.5 (range 1-22); 73% of patients remained progression-free at 12 weeks and 52% at 24 weeks. The predominant toxicity was uncomplicated myelosuppression: thrombocytopenia grade 3 (29%), grade 4 (10.4%); neutropenia grade 3 (12.5%), grade 4 (8.3%) anemia grade 3 (20.8%), grade 4 (4.2%). While pulmonary toxicity was reported, no patient had drug-related pneumonitis/hypoxia-type toxicity. Median progression-free survival (PFS) was 5.6+ months (range 0.7-27+ months). The median duration of objective response was 9+ months (range 3.9-24.5+ months). Fixed-dose rate gemcitabine plus docetaxel is active second-line therapy for uterine leiomyosarcoma.
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                Author and article information

                Journal
                Case Rep Oncol
                Case Rep Oncol
                CRO
                CRO
                Case Reports in Oncology
                S. Karger AG (Basel, Switzerland )
                1662-6575
                12 September 2023
                Jan-Dec 2023
                12 September 2023
                : 16
                : 1
                : 900-906
                Affiliations
                [a ]Minimally Invasive Gynecologic Surgery, University of California, Riverside, CA, USA
                [b ]Department of Internal Medicine, University of California, Riverside School of Medicine, Riverside, CA, USA
                Author notes
                Correspondence to: Austin R. Swisher, austin.swisher@ 123456medsch.ucr.edu

                Presentation: Abstract was presented virtually at the European Congress of Internal Medicine 2022 Meeting in Malaga, Spain.

                Article
                531761
                10.1159/000531761
                10601721
                37900811
                64233d0d-2c06-4f8d-a03f-791d94189a5d
                © 2023 The Author(s). Published by S. Karger AG, Basel

                This article is licensed under the Creative Commons Attribution 4.0 International License (CC BY) ( http://www.karger.com/Services/OpenAccessLicense). Usage, derivative works and distribution are permitted provided that proper credit is given to the author and the original publisher.

                History
                : 18 April 2023
                : 13 June 2023
                : 2023
                Page count
                Figures: 3, References: 20, Pages: 7
                Funding
                No funds, grants, or other support were received.
                Categories
                Case Report

                Oncology & Radiotherapy
                metastasis,uterine,leiomyosarcoma,thrombosis,embolism
                Oncology & Radiotherapy
                metastasis, uterine, leiomyosarcoma, thrombosis, embolism

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