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      Incidental deep soft tissue leiomyoma of the groin – a case report and comprehensive review of literature

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          Abstract

          Leiomyomas are rare, benign tumors arising from smooth muscle cells. Due to the heterogeneous anatomical locations, as well as clinical and radiological findings, early and correct diagnosis is usually tricky. The clinical symptoms depend on the anatomical structure the tumors are compromising by their growth. We present a case of a 53-year-old male patient who suffered from swelling of the right foot and ankle. Initially, arthritis and deep vein thrombosis were ruled out. As the swelling progressed to the entire leg, CT scan was performed showing a tumor compressing the right femoral vein. However, a magnetic resonance imaging (MRI) and a biopsy did not show any conclusive findings. Tumor excision was planned and immunohistochemical staining confirmed leiomyoma. The authors conclude that unspecific, progressive symptoms should prompt further investigation and whenever planning a biopsy, a possible malignant finding should be considered and excision of the biopsy canal together with the tumor must be assured.

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          Most cited references58

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          Hormonal manipulation of benign metastasizing leiomyomas: report of two cases and review of the literature.

          Benign metastasizing leiomyomas (BMLs) occur predominantly in women during reproductive years. The condition is characterized by uterine leiomyomas associated with the development, typically years later, of slow-growing metastatic lesions. The most commonly affected organs are the lungs, but BMLs have been reported in lymph nodes, deep soft tissues, mesentery, bones, the central nervous system, and the heart. In many cases, these lesions have an indolent course and are discovered rather incidentally. However, occasionally they can present with debilitating symptoms or even life-threatening complications. The presence of estrogen and progesterone receptors in these tumors supports their origin from uterine smooth muscle and, more importantly, makes them amenable to hormonal manipulation. Radical interventions, such as extensive tumor debulking and oophorectomy for hormonal control, although effective in many cases, are not always possible or desirable and carry significant morbidity. Here we present two cases of BMLs to illustrate the role of newer therapeutic agents, the estrogen receptor modulators and the aromatase inhibitors, in the hormonal manipulation of these tumors.
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            Solitary Cutaneous and Subcutaneous Leiomyoma

            A STOUT (1937)
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              Do leiomyomas of deep soft tissue exist? An analysis of highly differentiated smooth muscle tumors of deep soft tissue supporting two distinct subtypes.

              There is a prevailing view that leiomyomas of deep soft tissue are rare or nonexistent, but there are limited data on this subject in the form of large clinical studies with long follow-up information. We reviewed 36 consultation cases that had been diagnosed as leiomyoma or probable leiomyoma based on absence of nuclear atypia, necrosis, and no/minimal mitotic activity. Follow-up information was obtained to determine whether these stringent histologic criteria could identify a biologically benign group of smooth muscle tumors of deep soft tissue. The tumors occurred in two distinct locations. The first (n = 13) occurred in deep somatic soft tissue of the lower extremity (7), upper extremity (2), trunk (2), axilla (1), and back (1) and affected the sexes equally (7 male, 6 female). Composed of a circumscribed mass of mature smooth muscle cells, they were frequently calcified with a mean mitotic activity of <1 mitosis/50 high power fields (HPF) (range 1-4 mitoses/50 HPF). Estrogen receptor and progesterone receptor proteins were negative in the three cases tested. No tumors recurred or metastasized (mean follow-up 58.7 months, range 5-97 months). The second group (n = 23) occurred within the retroperitoneum (20) or abdominal cavity (3) of women (1 male, 22 female). Resembling uterine leiomyomas, they were always distinct from the uterus, occasionally multiple (n = 4), and sometimes occurred up to years after hysterectomy (n = 3). Four cases occurred with synchronous uterine leiomyomas. In the six cases tested, five of six were positive for the estrogen receptor protein and all were positive for progesterone receptor protein. Mean mitotic activity was 1 mitosis/50 HPF (range <1-10 mitoses/50 HPF). None developed metastasis within the follow-up period (mean 42.5 months, range 6-120 months); one tumor with a positive margin recurred at 10 months. We conclude that clinically benign smooth muscle tumors of deep soft tissue are rare but can be identified using stringent histologic criteria. They comprise two distinct subtypes: leiomyomas of somatic soft tissue and retroperitoneal-abdominal leiomyomas. The latter probably arise from hormonally sensitive smooth muscle. Although similar to uterine leiomyomas, they are located at sites removed from the uterus and are likely independent soft tissue primaries rather than parasitic leiomyomas of the uterus. We suggest that these two groups of smooth muscle tumors be diagnostically approached in a site-specific fashion.
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                Author and article information

                Contributors
                Journal
                J Surg Case Rep
                J Surg Case Rep
                jscr
                Journal of Surgical Case Reports
                Oxford University Press
                2042-8812
                January 2024
                30 January 2024
                30 January 2024
                : 2024
                : 1
                : rjae020
                Affiliations
                Department of Vascular Surgery, Cantonal Hospital Aarau , Tellstrasse 25, 5001 Aarau, Switzerland
                Department of Vascular Surgery, Cantonal Hospital Aarau , Tellstrasse 25, 5001 Aarau, Switzerland
                Gefässpraxis Dr. Allmann , Bahnhofstrasse 28, 6300 Zug, Switzerland
                Department of Pathology, Cantonal Hospital Aarau , Tellstrasse 25, 500 Aarau, Switzerland
                Department of Vascular Surgery, Cantonal Hospital Aarau , Tellstrasse 25, 5001 Aarau, Switzerland
                Author notes
                Corresponding author. Department of Vascular Surgery, Cantonal Hospital Sankt Gallen. E-mail: Falk_N@ 123456hotmail.com

                Falk Nessenius and Isabel Zucal share the co-first authorship of this work.

                Author information
                https://orcid.org/0009-0001-7994-0796
                Article
                rjae020
                10.1093/jscr/rjae020
                10832606
                38304319
                856b265d-da8f-49d2-a85f-74e603ee2f17
                Published by Oxford University Press and JSCR Publishing Ltd. © The Author(s) 2024.

                This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( https://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com

                History
                : 22 November 2023
                : 11 January 2024
                Page count
                Pages: 0
                Categories
                Case Report
                AcademicSubjects/MED00910
                jscrep/0130

                leiomyoma,lower extremity,tumor
                leiomyoma, lower extremity, tumor

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