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      A rare presentation of leiomyosarcoma metastasis to the cervical spine: A case report and a brief review

      case-report

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          Abstract

          Background:

          Leiomyosarcoma (LMS) is a rare malignancy that originates from smooth muscle. The most common sites of metastases include the lungs, liver, kidney, and skin. Notably, metastases of LMS to the central nervous system/or spine are extremely rare. When a cervical spinal LMS lesion was encountered, the patient successfully underwent gross total tumor resection with negative margins.

          Case Description:

          A 63-year-old female had undergone an anterior cervical C5–C7 diskectomy and fusion 18 years ago and resection of a retroperitoneal LMS 3 years ago. She newly presented with right-sided numbness and pain of 2 months duration that correlated with a focal right-sided C5-level hemiparesis (i.e., 4/5 motor strength). When the cervical magnetic resonance demonstrated a right-sided C5 intralaminar mass with extension into the C5–C6 foramen, she underwent posterior tumor resection; pathologically, this proved to be an LMS metastasis. Respectively, 1- and six months postoperatively, follow-up magnetic resonance imaging scans showed no tumor recurrence; she tolerated adjuvant oncological treatment accompanied by physical therapy. However, in one postoperative year, the lesion recurred, and she is presently under consideration for additional surgical management.

          Conclusion:

          Gross total surgical resection is the first line of treatment for patients with metastatic LMS. Here, a patient with a C5 laminar/C5–C6 foraminal bony LMS metastasis underwent posterior tumor resection accompanied by adjuvant oncological treatment but exhibited disease recurrence within one postoperative year.

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          Most cited references10

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          Incidence of Sarcoma Histotypes and Molecular Subtypes in a Prospective Epidemiological Study with Central Pathology Review and Molecular Testing

          Background The exact overall incidence of sarcoma and sarcoma subtypes is not known. The objective of the present population-based study was to determine this incidence in a European region (Rhone-Alpes) of six million inhabitants, based on a central pathological review of the cases. Methodology/Principal Findings From March 2005 to February 2007, pathology reports and tumor blocks were prospectively collected from the 158 pathologists of the Rhone-Alpes region. All diagnosed or suspected cases of sarcoma were collected, reviewed centrally, examined for molecular alterations and classified according to the 2002 World Health Organization classification. Of the 1287 patients screened during the study period, 748 met the criteria for inclusion in the study. The overall crude and world age-standardized incidence rates were respectively 6.2 and 4.8 per 100,000/year. Incidence rates for soft tissue, visceral and bone sarcomas were respectively 3.6, 2.0 and 0.6 per 100,000. The most frequent histological subtypes were gastrointestinal stromal tumor (18%; 1.1/100,000), unclassified sarcoma (16%; 1/100,000), liposarcoma (15%; 0.9/100,000) and leiomyosarcoma (11%; 0.7/100,000). Conclusions/Significance The observed incidence of sarcomas was higher than expected. This study is the first detailed investigation of the crude incidence of histological and molecular subtypes of sarcomas.
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            Leiomyosarcoma metastases to the spine. Case series and review of the literature.

            Leiomyosarcoma is a rare malignant smooth-muscle tumor that rarely metastasizes to bone. It is extremely uncommon for osseous metastasis to be the initial presentation of leiomyosarcoma or to be the initial manifestation of recurrence in patients with a history of leiomyosarcoma. The authors have treated four cases of metastatic leiomyosarcoma with the lesion initially presenting in the spine, and a fifth case of disseminated leiomyosarcoma that involved the spine. In their report, they highlight the cases of two of these patients and provide tabular data for the remaining three. The authors performed a comprehensive review of the literature on spinal leiomyosarcomas and retrospective chart reviews of five surgically treated patients in whom a spinal metastatic leiomyosarcoma was diagnosed. Their series consists of five women who ranged in age from 36 to 47 years (mean age 43.2 years). Four patients had known, or presumed, uterine primary lesions, whereas one harbored a retroperitoneal primary tumor. These lesions generally appear as lytic foci on imaging studies, but variable imaging characteristics were observed. All cases were managed aggressively: four patients underwent posterior/posterolateral decompression and fusion, and one underwent anterior-posterior en bloc resection and fusion. In all cases preoperative symptoms resolved. Two patients died 9 and 13 years after initial presentation. The remaining patients are alive and neurologically intact. Metastatic spinal leiomyosarcomas tend to symptomatically involve only one spinal level at the time of diagnosis and are known to recur locally. These lesions commonly affect women in early middle age, and long-term survival, even in those with systemic metastatic lesions, is better than that seen in individuals with more aggressive spinal metastases. Attempted gross-total resection with fusion, as opposed to minimal palliative decompression, is recommended.
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              Outcomes after surgery for spinal metastatic leiomyosarcoma.

              Leiomyosarcoma is a smooth-muscle sarcoma that rarely metastasizes to the spine. Its clinical course is variable, although patients with metastatic leiomyosarcoma can experience prolonged survival as compared with patients with more aggressive metastatic tumors. The authors report their single-institution experience in the surgical treatment of patients with leiomyosarcoma metastatic to the spine.
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                Author and article information

                Contributors
                Journal
                Surg Neurol Int
                Surg Neurol Int
                Surgical Neurology International
                Scientific Scholar (USA )
                2229-5097
                2152-7806
                2024
                12 April 2024
                : 15
                : 128
                Affiliations
                [1 ]Department of Neurosurgery, University Hospital Center Osijek, Osijek, Croatia
                [2 ]Faculty of Medicine, Josip Juraj Strossmayer University of Osijek, Osijek, Croatia
                [3 ]Department of Neurology, University Hospital Center Osijek, Osijek, Croatia
                [4 ]Department of Pathology and Forensic Medicine, University Hospital Center Osijek, Osijek, Croatia
                [5 ]Department of Oncology, University Hospital Center Osijek, Osijek, Croatia
                [6 ]Department of Diagnostic and Interventional Radiology, University Hospital Center Osijek, Osijek, Croatia
                [7 ]Department of Anesthesiology and Critical Care, University Hospital Center Osijek, Osijek, Croatia
                [8 ]Department of Neurology, Požega County Hospital, Požega, Požeško-Slavonska, Croatia.
                Author notes
                [* ] Corresponding author: Nenad Koruga, Department of Neurosurgery, University Hospital Center Osijek, Faculty of Medicine, Josip Juraj Strossmayer University of Osijek, Osijek, Croatia. nkoruga@ 123456gmail.com
                Article
                10.25259/SNI_66_2024
                10.25259/SNI_66_2024
                11090574
                38741997
                e07b192a-8a2d-4b7f-a3b9-48c2c0fffc29
                Copyright: © 2024 Surgical Neurology International

                This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-Share Alike 4.0 License, which allows others to remix, transform, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.

                History
                : 27 January 2024
                : 24 March 2024
                Categories
                Case Report

                Surgery
                leiomyosarcoma,metastasis,radiculopathy,spine
                Surgery
                leiomyosarcoma, metastasis, radiculopathy, spine

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