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      Benign metastasizing leiomyoma, a rare imposter of metastatic cervical cancer

      case-report

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          Highlights

          • Differential for necrotic pelvic mass with pulmonary nodules.

          • Benign metastasizing leiomyoma in woman with limited surgical history.

          • Efficacy of estrogen depletion therapy for benign metastasizing leiomyoma.

          Abstract

          Benign metastasizing leiomyoma (BML) is a rare variant of common benign smooth muscle neoplasm. We report a case of BML in a 45-year-old premenopausal woman with a significant smoking history and no previous history of cervical cancer screening. The patient presented with vaginal bleeding, abdominal pain, a necrotic mass in the vagina, and an incidental finding of diffuse pulmonary nodules on chest imaging. A total abdominal hysterectomy (TAH) and bilateral salpingo-oophorectomy (BSO) was performed for symptomatic management and adequate tissue sampling followed by fine needle biopsy of a pulmonary lesion. The pedunculated uterine mass and the pulmonary nodule were both consistent with smooth muscle neoplasm suggestive of myoma. Six-months following surgery, the patient is asymptomatic with partial regression of her lung nodules and no evidence of new or enlarging lesions while on treatment with Megestrol.

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          Most cited references10

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          Leiomyomas beyond the uterus: unusual locations, rare manifestations.

          Uterine leiomyomas affect 20%-30% of women older than 35 years. Extrauterine leiomyomas are rarer, and they present a greater diagnostic challenge: These histologically benign tumors, which originate from smooth muscle cells, usually arise in the genitourinary tract (in the vulva, ovaries, urethra, and urinary bladder) but may arise in nearly any anatomic site. In addition, unusual growth patterns may be seen, including benign metastasizing leiomyoma, disseminated peritoneal leiomyomatosis, intravenous leiomyomatosis, parasitic leiomyoma, and retroperitoneal growth. In the presence of such a pattern, a synchronous uterine leiomyoma or a previous hysterectomy for removal of a primary uterine tumor may be indicative of the diagnosis. However, some extrauterine leiomyomas may mimic malignancies, and serious diagnostic errors may result. The most useful modalities for detecting extrauterine leiomyomas are ultrasonography, computed tomography, and magnetic resonance (MR) imaging. The superb contrast resolution and multiplanar capabilities of MR imaging make it particularly valuable for characterizing these tumors, which usually show low signal intensity similar to that of smooth muscle on T2-weighted images. The radiologist's recognition of this and other characteristic features may help steer the clinician toward timely, appropriate management and away from unnecessary, potentially harmful treatment.
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            Uterine fibroids: current perspectives

            Uterine fibroids are a major cause of morbidity in women of a reproductive age (and sometimes even after menopause). There are several factors that are attributed to underlie the development and incidence of these common tumors, but this further corroborates their relatively unknown etiology. The most likely presentation of fibroids is by their effect on the woman’s menstrual cycle or pelvic pressure symptoms. Leiomyosarcoma is a very rare entity that should be suspected in postmenopausal women with fibroid growth (and no concurrent hormone replacement therapy). The gold standard diagnostic modality for uterine fibroids appears to be gray-scale ultrasonography, with magnetic resonance imaging being a close second option in complex clinical circumstances. The management of uterine fibroids can be approached medically, surgically, and even by minimal access techniques. The recent introduction of selective progesterone receptor modulators (SPRMs) and aromatase inhibitors has added more armamentarium to the medical options of treatment. Uterine artery embolization (UAE) has now been well-recognized as a uterine-sparing (fertility-preserving) method of treating fibroids. More recently, the introduction of ultrasound waves (MRgFUS) or radiofrequency (VizAblate™ and Acessa™) for uterine fibroid ablation has added to the options of minimal access treatment. More definite surgery in the form of myomectomy or hysterectomy can be performed via the minimal access or open route methods. Our article seeks to review the already established information on uterine fibroids with added emphasis on contemporary knowledge.
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              Benign metastasizing leiomyoma: A review of current literature in respect to the time and type of previous gynecological surgery

              Introduction Benign metastasizing leiomyoma (BML) is a rare disorder that affects women with a history of uterine leiomyoma, which is found to metastasise within extrauterine sites. The aetiology of BML remains unexplained. Because BML is rare, and most publications contain descriptions of single cases, no statistically determined time relations were found between the primary and secondary surgeries, which may have aetiological implications. Objectives To determine age before BML surgery, age during diagnosis of BML, type of prior surgery, and location of metastasis based on the literature. Methods A systematic review of four databases (Medline/PubMed, Embase, Web of Science, and Cochrane) covering articles published from 1 January 1965 to 10 April 2016. The inclusion criteria were full-text articles in English and articles containing case reports. Articles in languages other than English (39), articles containing incomplete data (14), i.e. no information regarding the time of surgery and/or the site of metastasis, articles bereft of case studies (25), and articles with access only to summaries, without access to the complete text (10) were excluded. Of 321 titles identified, only 126 articles met the aforementioned criteria. Results and conclusions The mean age during primary surgery and BML diagnosis was 38.5 years and 47.3 years, respectively. The most common surgery was total hysterectomy. The most frequent site of metastasis was the lungs; other organs were affected less frequently.The site of metastases and their number were not related to the longer time span between the patient’s initial surgery and occurrence of metastasis. The analysed data, such as the age during primary surgery, age during BML diagnosis, site and type of metastasis, do not provide us a clear answer. Thus, BML pathogenesis is most probably complex in nature and requires further multidirectional research.
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                Author and article information

                Contributors
                Journal
                Gynecol Oncol Rep
                Gynecol Oncol Rep
                Gynecologic Oncology Reports
                Elsevier
                2352-5789
                20 November 2021
                November 2021
                20 November 2021
                : 38
                : 100893
                Affiliations
                [a ]Elson S Floyd College of Medicine, Washington State University, Spokane, WA, United States
                [b ]Compass Oncology, United States
                [c ]Society of Gynecologic Oncology, United States
                Author notes
                [* ]Corresponding author. stella.whang@ 123456wsu.edu
                Article
                S2352-5789(21)00197-1 100893
                10.1016/j.gore.2021.100893
                8651751
                34926773
                d08af96c-3dfa-4e8a-a096-aa2eed2a1206
                © 2021 The Authors. Published by Elsevier Inc.

                This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

                History
                : 30 July 2021
                : 13 November 2021
                : 16 November 2021
                Categories
                Case Report

                benign metastasizing leiomyoma,prolapsing leiomyoma,pulmonary nodules

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