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      Concurrent benign metastasizing leiomyoma in the lung and lumbar spine with elevated standardized uptake value level in positron-emission tomography computed tomography : A case report and literature review

      case-report
      , MD, , MD, , MD, , PhD, , PhD, , PhD
      Medicine
      Wolters Kluwer Health
      benign metastasizing leiomyoma, lung, metastasis, spine, uterine leiomyoma

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          Abstract

          Rationale:

          Benign metastasizing leiomyoma (BML) is rare condition involving distant metastases secondary to benign uterine leiomyoma, and it is most commonly found in the lungs. It rarely metastasizes to the spine to cause osteolytic damage and spinal canal compression.

          Patient concerns:

          A 51-year-old woman with low back and bilateral leg pain and paresthesia was admitted to our ward. She has a previous medical history of uterine leiomyomas. Magnetic resonance imaging of the lumbar spine revealed vertebral body osteolytic destruction and soft tissue mass in the L4/5 with a secondary lumbar spinal stenosis. Positron emission tomography computed tomography showed moderately intense accumulation of 18F-fluorodeoxyglucose in the L4/5 mass, as well as multiple nodules with increased metabolic activity in both lungs.

          Diagnoses:

          Pulmonary and spinal BML.

          Interventions:

          The patient underwent a computed tomography-guided percutaneous needle biopsy of the lung nodule and lumbar corpectomy, tumor excision, and vertebroplasty in the L4/5.

          Outcomes:

          Pathologically, both pulmonary nodule and vertebral mass were diagnosed as leiomyomas without any malignant evidence. Estrogen and progesterone receptors were both positive in the metastatic tumors. The patient's symptoms completely disappeared after the surgery. The patient is currently receiving outpatient anti-estrogen tamoxifen treatment for a BML.

          Lessons:

          Through this case, we suggest that BML should be regarded as part of differential diagnosis in female patients with a previous medical history of uterine leiomyomas presenting with multiple nodules in any parts of the body.

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          Most cited references24

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          Pulmonary benign metastasizing leiomyoma: a case report and literature review.

          Benign metastasizing leiomyoma (BML) is a rare condition that occurs in all age groups and that is particularly prevalent among women of late childbearing age. All patients have a history of uterine leiomyoma and/or myomectomy, often associated with distant metastases from the uterus, which commonly occurs in the lung. We report the case of a 32-year-old young woman suffering from chest stuffiness, labored respiration and weakness after a myomectomy performed one month earlier. The chest CT showed a diffuse miliary shadow in both sides of her lungs, but serum tumor markers such as CA125, CA199, carcinoembryonic antigen (CEA), neuron specific enolase (NSE), and CYFRA21-1 were normal. The patient underwent a lung biopsy by thoracoscopic surgery after four weeks of anti-TB treatment; there were no significant changes in the chest CT. H&E staining showed that the tumor cells had characteristics of smooth muscle cell differentiation. Immunohistochemical staining showed a low tumor cell proliferation index, which indicated that the likelihood of a malignancy was not high. There was no expression of CD10, indicating a diagnosis of pulmonary benign metastasizing leiomyoma (PBML). Smooth muscle actin (SMA) and desmin as specific markers of smooth muscle and the estrogen receptor (ER) and progesterone receptor (PR) were all strongly positive, which is characteristic of PBML. The patient was given the anti-estrogen tamoxifen for 3 months. With no radiological evidence of disease development and further distant metastasis, the patient will continue to be followed.
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            Pulmonary benign metastasizing leiomyoma associated with intravenous leiomyomatosis of the uterus: clinical behavior and genomic changes supporting a transportation theory.

            Benign metastasizing leiomyoma is a rare lesion characterized by benign-appearing smooth muscle tumor most frequently involving the lung and usually associated with a benign leiomyoma or intravenous leiomyomatosis of the uterus. The pathogenetic mechanism of the tumor has not been clarified, but the possibilities including hormone-sensitive in situ proliferations of smooth muscle bundles, mechanical displacement or intravascular spread of preexisting benign uterine tumor tissue, and metastasized very low-grade uterine leiomyosarcoma have been proposed. We described a case of pulmonary benign metastasizing leiomyoma associated with a uterine intravenous leiomyomatosis in a 46-year-old woman with a result of comparative genomic hybridization study. The 2 lesions showed significantly overlapping, if not identical, complex genomic changes in the comparative genomic hybridization, suggesting that the 2 lesions are closely related to each other. Unresected pulmonary nodules were left untreated for 13 months after the hysterectomy and wedge biopsy of 3 pulmonary nodules to show no further growth, suggesting clinical behavior of nonmalignant tumor in our case. Benign metastasizing leiomyomas may comprise a heterogeneous group of tumors in terms of their malignant potential and pathogenetic mechanism.
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              Benign metastasizing leiomyoma of the lung

              Benign leiomyomas of the uterus are uncommonly found in association with benign smooth muscle tumors beyond the confines of the uterus. Benign metastasizing leiomyoma (BML) is a rare disease in which the lung is described to be the most afflicted extrauterine organ. We present a brief review of the literature, along with case reports for four patients who were followed up after resection of a pulmonary lesion or after pathological confirmation by biopsy. The clinical course of BML varies from chronic asymptomatic appearance to rapid progression, leading to respiratory failure and death. Our BML patients did not complain of pulmonary symptoms, such as cough, dyspnea, or chest tightness. Pathology revealed benign leiomyomas with no atypia and mitotic activity <5 per 10 high-power field. Immunohistochemical staining was positive for actin and desmin. A standard treatment for BML has not yet been established. Because of the hormone-sensitive characteristics of BML, treatments are based on hormonal manipulation along with either surgical or medical oophorectomy. Benign metastasizing leiomyoma can be observed in postmenopausal women. We observed four patients who did not receive adjuvant hormonal therapy because they were postmenopausal or perimenopausal. All patients are still healthy and show no evidence of recurrence or progression of the disease.
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                Author and article information

                Journal
                Medicine (Baltimore)
                Medicine (Baltimore)
                MEDI
                Medicine
                Wolters Kluwer Health
                0025-7974
                1536-5964
                July 2018
                06 July 2018
                : 97
                : 27
                : e11334
                Affiliations
                Department of Respiratory Medicine, the Second Xiangya Hospital, Research Unit of Respiratory Disease, Diagnosis and Treatment Center of Respiratory Disease, Central South University, Changsha, China.
                Author notes
                []Correspondence: Ruoyun Ouyang, Department of Respiratory Medicine, the Second Xiangya Hospital, Research Unit of Respiratory Disease, Diagnosis and Treatment Center of Respiratory Disease, Central South University, Changsha, Hunan 410011, China (e-mail: ouyangruoyun@ 123456csu.edu.cn ).
                Article
                MD-D-18-01243 11334
                10.1097/MD.0000000000011334
                6076117
                29979408
                ba28194e-d3b1-46ca-8403-18c01f8c6be1
                Copyright © 2018 the Author(s). Published by Wolters Kluwer Health, Inc.

                This is an open access article distributed under the terms of the Creative Commons Attribution-Non Commercial-No Derivatives License 4.0 (CCBY-NC-ND), where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal. http://creativecommons.org/licenses/by-nc-nd/4.0

                History
                : 16 March 2018
                : 28 May 2018
                Categories
                7400
                Research Article
                Clinical Case Report
                Custom metadata
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                benign metastasizing leiomyoma,lung,metastasis,spine,uterine leiomyoma

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