Primary leiomyosarcoma of the thoracic outlet: case report and surgical approach

Introduction

Background

Leiomyosarcoma is a malignant neoplasm of smooth muscle origin, accounting for approximately 10–25% of soft tissue sarcomas (1-3). These tumors most frequently present between 40 and 60 years of age with a slight female predominance (1,4-6). Leiomyosarcomas most commonly occur in the retroperitoneum and uterus, and only rarely arise in intravascular sites (7-9). Imaging studies are essential to characterize the mass and its anatomical relationships for surgical planning, but core needle or excisional biopsy remains the diagnostic gold standard (10,11). Whole body imaging is required in the diagnostic workup because higher-grade lesions frequently metastasize (10,12).

Poor prognostic factors for leiomyosarcoma include high histologic grade, large tumor size, and presence of metastases at diagnosis (5,10,13,14). Management is determined by lesion size, location, depth, grade, and patient-specific factors (4,7,10). Complete surgical resection with negative margins (R0 resection) is the only intervention consistently associated with improved survival outcomes (7,10,15). Systemic therapy is reserved for select high-grade or unresectable cases (4,7,10,16). Following initial treatment, long-term surveillance with clinical examination and imaging is recommended due to the risk of recurrence and metastases (10).

Rationale and knowledge gap

Leiomyosarcoma of the thoracic outlet is exceedingly rare, with no standardized surgical approach established for resection. Thoracic outlet tumor resections of other cell origins have been described as highly challenging due to their deep location and close proximity to multiple neural and vascular structures, including the phrenic, vagus, and recurrent laryngeal nerves, supra-aortic arterial branches, and major venous structures such as the subclavian vein and brachiocephalic trunk (17-20). When thoracic outlet tumors involve major veins, surgical complexity increases due to venous wall adherence requiring en bloc resection and vascular reconstruction (21,22). Furthermore, as complete surgical resection with negative margins is the only potentially curative treatment, vascular leiomyosarcoma often requires multidisciplinary collaboration between surgical oncology and vascular surgery (23,24).

Most published cases on thoracic outlet tumors have utilized traditional surgical techniques, including claviculectomy, supraclavicular, or infraclavicular approaches (17,18). These techniques may provide inadequate tumor exposure for more medially positioned or posteriorly involving tumors (19,20). The transmanubrial-transclavicular approach described by García-López and Iborra for resection of brachial plexus fibromatoses offers significantly improved exposure of the thoracic outlet, potentially enhancing complete tumor resection while minimizing damage to surrounding structures (19,20).

Objective

Herein, we report a patient with thoracic outlet leiomyosarcoma involving the left subclavian vein who underwent complete surgical resection via the transmanubrial-transclavicular approach. To our knowledge, this is the first reported surgical management of thoracic outlet leiomyosarcoma involving the left subclavian vein. By sharing our experience and operative strategy, we aim to provide a framework for the management of this rare and anatomically challenging presentation. We present this article in accordance with the CARE reporting checklist (available at https://ccts.amegroups.com/article/view/10.21037/ccts-2025-1-52/rc).

Case presentation

A 68-year-old woman with no significant past medical history presented with a painless left neck mass, initially noted 4 years ago and dismissed by the patient as a benign cyst. She denied any systemic symptoms, including fevers, night sweats, or weight loss. Her family history was notable for stomach cancer, colon cancer, and melanoma, with no known history of sarcoma or hereditary cancer syndromes. The mass slowly enlarged over time, and she later developed fatigue and intermittent left-sided swelling in her arm and hand, which was clinically attributed to venous obstruction. Approximately 2 years after the mass first appeared, she developed left subclavian deep vein thrombosis and was treated with percutaneous thrombectomy and dilation of the left subclavian vein. She was subsequently placed on direct oral anticoagulation and referred for further diagnostic evaluation.

On physical examination, there was a firm, palpable mass in the left supraclavicular region, limited passive range of motion in the left upper extremity, but no focal neurologic deficits. Laboratory evaluation was notable for a mild leukocytosis without clinical evidence of infection. Computed tomography (CT) angiography and magnetic resonance imaging (MRI) of the chest and neck identified a tubular, enhancing soft tissue mass (9 cm × 3 cm) partially occluding the left subclavian vein with superior extension into the left lower neck region, prominent venous collateralization, and no cervical adenopathy (Figures 1,2). Although the mass was anatomically adjacent to neural structures within the thoracic outlet, fat-saturated T2-weighted MRI demonstrated no evidence of brachial plexus compression (Figure 3). Whole-body positron emission tomography (PET)/CT demonstrated that the mass had hypermetabolic activity (standardized uptake value max 6.2) (Figure 2), with no evidence of a primary tumor at more common sites, including the uterus or retroperitoneum, and a right upper lobe pulmonary nodule that was too small for accurate PET characterization, but no clear evidence of metastatic disease. Duplex venous ultrasound showed that the mass was vascularized, and that the left upper extremity veins were patent without evidence of deep vein thrombosis (Figure S1). CT-guided core needle biopsy of the neck mass was obtained, with immunohistochemical (IHC) staining diffusely positive for smooth muscle actin (SMA) and desmin, supportive of a smooth muscle phenotype (Figure S2). There was sparse mitotic activity, a moderate degree of tumor cell pleomorphism, and no tumor necrosis, consistent with a diagnosis of leiomyosarcoma, preliminary Fédération Nationale des Centres de Lutte Contre le Cancer (FNCLCC) grade I. In the absence of another identifiable primary site on whole-body imaging, the lesion was most consistent with a primary leiomyosarcoma of the thoracic outlet arising from the left subclavian vein. Molecular profiling was not performed given the definitive histopathologic diagnosis established by morphology and immunohistochemistry and the absence of metastatic disease, consistent with current guidelines recommending selective use of gene sequencing in sarcomas (10,25).

Figure 1 Coronal (A) and sagittal (B) chest CT angiography showing a tubular mass in the left supraclavicular region extending into the left subclavian vein (arrows). CT, computed tomography.

Figure 2 Axial (A,B) and coronal (C) fat-saturated post-contrast T1-weighted MRI images demonstrating an enhancing, tubular mass in the left supraclavicular region extending into the left subclavian vein (arrows). (D) The enhancing mass corresponds to an FDG-avid lesion (arrow) in the coronal PET-CT image. FDG, fluorodeoxyglucose; MRI, magnetic resonance imaging; PET-CT, positron emission tomography-computed tomography.

Figure 3 Coronal fat-saturated T2-weighted MRI image demonstrating a hyperintense mass (red arrows) that is separate from the nerve trunks of the brachial plexus (green arrows). MRI, magnetic resonance imaging.

Her case, imaging, and pathology were discussed at a multidisciplinary sarcoma tumor board, with consensus on a diagnosis of grade I leiomyosarcoma of the left subclavian vein, extending proximally to the external jugular vein and distally to the start of the left axillary vein. Systemic therapy is not indicated for grade I leiomyosarcoma, and she was scheduled for tumor resection with the thoracic and vascular surgery teams. The patient’s direct oral anticoagulant was held 72 hours prior to surgery.

Intraoperative assessment demonstrated that conventional thoracic outlet exposure was insufficient to achieve safe vascular control due to dense tumor adherence to the left subclavian vein and greater medial extension toward the internal jugular vein and venous confluence than anticipated based on preoperative imaging. She subsequently underwent successful R0 resection via partial claviculectomy and partial sternotomy (Figure 4), with left subclavian vein resection and reconstruction, as described in detail below.

Figure 4 Schematic of the surgical approach outlining the extent of the partial sternotomy and partial claviculectomy (dashed lines). The relationship between the mass and the surrounding structures is also shown.

An incision superior to the left clavicle was carried down to the subplatysmal space, and the mass was encountered less than 2 cm below the skin layer and 2 cm above the clavicle. The mass was well encapsulated, requiring meticulous dissection to preserve uninvolved vein and nerve branches. The lateral margin of the mass terminated at the distal subclavian vein, which was identified and controlled with a vessel loop. To adequately dissect out the medial and inferior tumor borders, the middle third of the clavicle and the sternoclavicular joint were excised. With the clavicle removed, the subclavian vein was identified to be densely adherent to the mass. The tumor extended deep and medially to the level of the thoracic outlet, involving the venous confluence. During deep dissection of the medial plane, the clavicular head of the sternocleidomastoid was divided, and the scalene fat pad was entered, with preservation of the phrenic nerve. The left external jugular vein was ligated cephalad to the tumor due to tumor involvement in the more caudal portion. The medial aspect of the tumor could not initially be visualized, necessitating a partial sternotomy to further expose the venous confluence. With full exposure, it was evident that the tumor involved the subclavian vein with extension to the venous confluence of the subclavian and internal jugular veins, but a clean uninvolved plane was identified at the confluence for proximal resection (Figure 5). The innominate, left internal jugular, and left internal mammary veins were isolated and controlled. The tumor was freed from all soft tissue attachments until only the segment involving the left subclavian vein remained. No lymphadenopathy was identified intraoperatively, and formal lymph node dissection was not performed. The patient then received 10,000 units of intravenous heparin to achieve an activated clotting time of approximately 250 seconds. The innominate and internal jugular veins were clamped proximally, and the distal subclavian vein was clamped distally, allowing for resection of the section of subclavian vein with tumor involvement. The specimen was resected with margins negative for malignancy on frozen section. An 8 mm ringed polytetrafluoroethylene (PTFE) graft was fashioned as an interposition graft between the distal subclavian and innominate veins, preserving the inflow from the jugular vein. Antegrade and retrograde bleeding was performed before completing the final anastomosis and releasing clamps to allow for de-airing of the PTFE graft. The patient was reversed with protamine. The subclavian vein and innominate veins were auscultated with a Doppler probe and found to have excellent, continuous flow. The sternum was reapproximated and two channel drains were left in the substernal and supraclavicular spaces. The patient tolerated the procedure well without any immediate complications.

Figure 5 Intraoperative images from the surgeon’s perspective (patient’s left side with the patient’s head to the right and the patient’s feet to the left). Surgical approach via (A) partial sternotomy (dashed line) and partial claviculectomy (dashed box) is shown. (B) The mass (dashed outline) is shown relative to the ligated venous structures.

On gross pathologic assessment, the tumor appeared confined to the lumen of the resected veins and measured 11.0 cm in greatest dimension (along the length of the subclavian vein) (Figure 6). The tumor was solid and firm with homogeneous tan-white coloration on cut sections. Microscopic sections confirmed the gross impression of an entirely intravascular tumor (Figure S3), which comprised a moderately cellular, pleomorphic spindle cell neoplasm with fascicular architecture and focal tumor necrosis (FNCLCC grade II) (Figure S4). The tumor cells had slightly enlarged, elongated nuclei with blunt ends and fibrillary eosinophilic cytoplasm, indicative of smooth muscle differentiation (Figure S4).

Figure 6 The 11.0-cm resected specimen involving the left subclavian and innominate veins, shown (A) in the operating room and (B) on gross pathologic examination.

In the immediate postoperative period, she was noted to have a chyle leak through her supraclavicular drain which improved with a no-fat diet modification. Given the clavicular resection, the patient was provided with a shoulder sling and participated in early inpatient physical therapy. On postoperative day 5, she was discharged on her home direct oral anticoagulant, newly started aspirin 81 mg daily, a short course of oxycodone, and a 2-week course of trimethoprim-sulfamethoxazole. The drains were removed during her postoperative follow-up appointment. Continued surveillance with medical oncology was recommended given successful R0 resection of a grade II leiomyosarcoma of the left subclavian vein. In this case, adjuvant radiotherapy was not pursued given the margin-negative resection, the technical challenges of delivering radiation to the thoracic outlet without excessive morbidity to surrounding neurovascular structures, and patient preference following multidisciplinary discussion. At 3-month follow-up, the patient reported overall good recovery, with mild left upper extremity swelling, occasional arm fatigue, and intermittent incisional pain. Surveillance imaging showed no evidence of recurrence or metastatic disease, with repeat imaging planned in 4 months. All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration and its subsequent amendments. Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

Discussion

Key findings

We present a case in which a thoracic outlet leiomyosarcoma involving the left subclavian vein was successfully resected via the transmanubrial-transclavicular approach. To our knowledge, this is the first documented application of this surgical technique to leiomyosarcoma in the thoracic outlet region. By mobilizing the medial clavicle, a portion of the sternal manubrium, and the sternocleidomastoid muscle, this method provided exceptional operative exposure and facilitated careful dissection around critical neurovascular structures allowing for complete tumor excision and venous reconstruction. This experience supports the utility of such technique for managing similarly challenging thoracic outlet tumors.

Strengths and limitations

A primary strength of this report is the successful management of a rare, anatomically complex tumor using a non-traditional surgical approach that provided excellent exposure and adequate control of critical structures. However, the findings are limited to a single case and a short duration of follow-up, which restricts broader conclusions about long-term oncologic outcomes and reconstructive durability. Late tumor recurrence or graft-related complications may still occur and cannot be excluded. In addition, the need for specialized surgical expertise and multidisciplinary support may limit the generalizability of this approach to all treatment centers. We recommend establishing early referral pathways for thoracic outlet tumors to facilitate timely access to appropriate care.

Comparison with similar research

Most reported cases of thoracic outlet tumors are benign, including schwannomas, neurofibromas, and fibromatoses (26-28). Malignant tumors, including synovial sarcoma, malignant nerve sheath tumors, and myxofibrosarcoma, have also been reported in the thoracic outlet (27,29-33). Benign and select malignant thoracic outlet tumors are typically managed with traditional surgical approaches such as supraclavicular, infraclavicular, and claviculectomy (17,18,28). These standard exposures are generally adequate for superficial or less infiltrative lesions but may provide inadequate exposure for more medially positioned, deeply situated, or invasive tumors, particularly those that encase or attach to major neurovascular structures (19,20).

Malignant thoracic outlet tumors with deep extension or neurovascular involvement often require more advanced surgical approaches to facilitate vascular control and en bloc resection. These include the anterior transclavicular (Dartevelle) and transmanubrial osteomuscular sparing (Grunenwald) approaches (34). In these cases, adequate exposure is critical, as complete surgical resection with negative margins has consistently been identified as the primary determinant of long-term oncologic outcomes, whereas incomplete resection is associated with poor survival (29,30,35).

The transmanubrial-transclavicular approach, originally for benign tumors in the thoracic outlet, offers enhanced exposure of the thoracic outlet but has not previously been reported for resection of thoracic outlet leiomyosarcoma (20). By introducing this approach for thoracic outlet leiomyosarcoma involving the subclavian vein, we highlight a new exposure option for challenging malignant tumors of the thoracic outlet when conventional techniques are insufficient to achieve complete resection.

Explanations of findings

Traditional surgical approaches may limit access to critical thoracic outlet structures, theoretically increasing the risk of incomplete resection or iatrogenic injury. In this case, long-segment circumferential subclavian vein involvement extending into the venous confluence and near the internal jugular vein precluded the use of single approaches typically utilized for thoracic outlet tumors. The combined transmanubrial and transclavicular exposure allowed for careful and precise dissection around the subclavian vein, safe en bloc tumor resection with R0 margins, and venous reconstruction within the confined thoracic outlet. Our result also emphasizes the value of detailed preoperative imaging, multidisciplinary collaboration, and an individualized operative strategy.

Implications and actions needed

While this single case cannot establish the superiority of the transmanubrial-transclavicular approach, it demonstrates its feasibility for thoracic outlet leiomyosarcoma and may inform surgical planning in similar anatomic scenarios. Wider reporting of similar cases, along with documentation of outcomes, will be essential to the development of best-practice protocols for tumors located within the thoracic outlet. For patients with comparable clinical scenarios, referral to centers with multidisciplinary oncologic and advanced surgical expertise is recommended.

Conclusions

This case demonstrates the feasibility and safety of the transmanubrial-transclavicular approach for the resection of a thoracic outlet leiomyosarcoma involving the subclavian vein. When conventional approaches provide insufficient exposure, the transmanubrial-transclavicular technique should be considered for resection of select thoracic outlet tumors. Advanced imaging, referral to specialized centers, and multidisciplinary surgical planning are also recommended. Broader reporting and long-term follow-up of similar cases will be necessary to define best practices for the management of rare sarcomas of the thoracic outlet.

Acknowledgments

None.

Footnote

Reporting Checklist: The authors have completed the CARE reporting checklist. Available at https://ccts.amegroups.com/article/view/10.21037/ccts-2025-1-52/rc

Peer Review File: Available at https://ccts.amegroups.com/article/view/10.21037/ccts-2025-1-52/prf

Funding: None.

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://ccts.amegroups.com/article/view/10.21037/ccts-2025-1-52/coif). C.N.E. received speaker honoraria from AtriCure and consultation fees from Johnson & Johnson MedTech and Cook Medical (expired). The other authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration and its subsequent amendments. Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

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