Neck subcutaneous nodule as first metastasis from broad ligament leiomyosarcoma: a case report and review of literature
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CASE REPORT
Neck subcutaneous nodule as first metastasis from broad ligament leiomyosarcoma: a case report and review of literature Fiorella Cazzato1, Angela D’Ercole2, Graziano De Luca3, Francesca B. Aiello4 and Adelchi Croce1*
Abstract Background: Leiomyosarcoma usually develops in the myometrium and is characterized by a high recurrence rate, frequent hematogenous dissemination, and poor prognosis. Metastasis is usually to lungs, liver, and bone, and occasionally to the brain, but seldom to the head and neck region. Primary leiomyosarcoma very rarely arises in the broad ligament. Case presentation: A 54-year old woman presented to the otolaryngology department with a mass in the right posterior region of the neck 4 years after surgery for a primary leiomyosarcoma of the right broad ligament. The neck mass was removed and found to be a metastatic leiomyosarcoma. Leiomyosarcoma localizations in lungs and liver were absent. Morphological examination showed both the primary and the secondary leiomyosarcomas to have features of low-grade tumors. One year after excision of the neck mass, the patient presented with tachycardia. Echocardiography detected two intracardiac nodules suggestive of metastatic tumors. Chemotherapy was administered; the disease has been stable since then. Conclusions: We report the first case of broad ligament leiomyosarcoma with the neck subcutaneous region being the first site of secondary involvement. We speculate that the Batson venous plexus might have been the pathway of dissemination. Keywords: Broad ligament leiomyosarcoma, Head and neck leiomyosarcoma, Atypical uterine smooth muscle tumors, Metastasis, Batson plexus, Case report Background Leiomyosarcoma is a malignant tumor derived from smooth muscle [1]. It is most common in women in the fifth decade of life, with the myometrium being the usual site of origin [2]. These tumors accounts for 1% of all gynecological malignancies, 3.7% of uterine malignant tumors, and 25–36% of uterine sarcomas [3]. The *Correspondence: [email protected] 1 Department of Medical, Oral and Biotechnological Sciences, University G. D’Annunzio, Chieti‑Pescara, Via dei Vestini, 66100 Chieti, Italy Full list of author information is available at the end of the article
incidence of uterine leiomyosarcoma is 0.36 per 100,000 women–years [4]. Leiomyosarcoma of the broad ligament is very rare, with only 24 cases reported to date [5]. Gynecological leiomyosarcoma has poor prognosis because of local invasiveness and a tendency for distant metastasis usually to the liver, lung, and bone [6]. Metastasis to the head and neck region is rare, with only 24 cases reported in the literature [2, 7–12]. The rarity of metastasis to the head and neck increases the likelihood of misdiagnosis or delayed diagnosis [1, 13], particularly when concurrent liver or lung metastases are absent. We report a very rare case of a primary leiomyosarcoma in
© The Author(s) 2020. Open Access This article is licensed under a Creative Commons Attribution 4.0 International License
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