Neck subcutaneous nodule as first metastasis from broad ligament leiomyosarcoma: a case report and review of literature

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: Atypical uterine smooth muscle tumors; Batson plexus; Broad ligament leiomyosarcoma; Case report; Head and neck leiomyosarcoma; Metastasis.

Fig. 1

Representative preoperative, CT scan, MRI, and surgical excision images. a Preoperative photograph of the mass in the right posterior neck region. b CT scan showing a solid mass (indicated by the arrow) in the posterior neck region near C1-C2. c MRI image showing a hyper-intense mass displacing paravertebral muscles (arrow), and a cleavage plane between the mass and the sternocleidomastoid muscle. d Surgical excision of the mass by posterior neck cervicotomy

Fig. 2

Immunohistochemical evaluation of broad ligament and neck leiomyosarcomas. a MIB-1 immunoreactivity in the primary LMS. b MIB1 immunoreactivity in the neck LMS. Insets in a and b show correspondent hematoxylin–eosin stainings. c, Smooth muscle actin immunoreactivity in the primary LMS. d, Smooth muscle actin immunoreactivity in the neck LMS. e, Desmin immunoreactivity in the primary LMS. f, Desmin immunoreactivity in the neck LMS. G, p16 immunoreactivity in the primary LMS. h, p16 immunoreactivity in the neck LMS. (a–h: × 200)

Fig. 3

Chest CT scan (July 2020). Intracardiac masses exhibiting inhomogeneous intensity. a Nodule on the lateral wall of the left ventricle (arrow). b Nodule at the apex of the right ventricle (arrow)