Ann Thorac Surg 2002;74:567-569
© 2002 The Society of Thoracic Surgeons
Case report
Mediastinal epithelioid hemangioendothelioma resected by hemi-plastron window technique
Noritaka Isowa, MDa,
Seiki Hasegawa, MDa,
Mari Mino, MDb,
Kojiro Morimoto, MDc,
Hiromi Wada, MD*a
a Department of Thoracic Surgery, Graduate School of Medicine, Kyoto University, Kyoto, and Japan
b Department of Pathology, Graduate School of Medicine, Kyoto University, Kyoto, and Japan
c Department of Thoracic Surgery, Kyoto City Hospital, Kyoto, Japan
Accepted for publication April 1, 2002.
* Address reprint requests to Dr Wada, Department of Thoracic Surgery, Graduate School of Medicine, Kyoto University, 54 Shogoin Sakyo-ku, Kyoto 606-8507, Japan
e-mail: wadah{at}kuhp.kyoto-u.ac.jp
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Abstract
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We report a case of epithelioid hemangioendothelioma arising from the left brachiocephalic vein in the anterior superior mediastinum of a 41-year-old man. The tumor was resected by local excision using a novel approach, the "hemi-plastron window" technique. The patient had an uneventful postoperative course and no recurrence of tumor 28 months after surgical treatment. We describe this surgical approach and discuss its advantage for cervicothoracic tumors.
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Introduction
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Epithelioid hemangioendothelioma is a rare epithelial-appearing vascular tumor, displaying a clinical behavior intermediate between that of hemangioma and angiosarcoma [1]. This is a case report of mediastinal epithelioid hemangioendothelioma.
During routine mass screening examinations, a 41-year-old man underwent a chest roentgenogram revealing a left superior anterior mediastinal mass (Fig 1A),
and was referred to our hospital for further examination. A chest roentgenogram taken 3 years earlier detected no abnormal shadow (Fig 1B). His medical and family histories and clinical status were unremarkable. A complete hematological examination, including tumor markers such as carcinoembryonic antigen, cytokeratin fragment 21, alpha-fetoprotein, carbohydrate antigen 19-9, and sialyl Lewis (x) antigen, were all negative. A computed tomographic scan of the chest demonstrated a heterogenous mass (34 mm x 27 mm) with calcification. Magnet resonance imaging revealed that the mediastinal mass was located lateral to the left common carotid artery and anterior to the left subclavian artery (Fig 2).
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Fig 1. (A) Superior anterior mediastinal mass (arrow) is shown on the chest roentgenogram on admission. (B) Chest roentgenogram, taken 3 years before admission, reveals no abnormal shadow.
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Fig 2. Magnetic resonance images. (A) Contrast-enhanced T1-weighted image shows a well-enhanced mass between the left common carotid artery (arrow) and the subclavian artery (arrowhead). (B) T2-weighted image with fat suppression shows a soft tissue mass of heterogeneous signal intensity.
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Operation was performed on October 12, 1999. In the supine position, the skin was incised along the upper border of left clavicle from the midclavicular line to the suprasternal notch. It was continued vertically in the midsternal plane to the second intercostal space. After resection of 3 cm of the medial portion of the left clavicle, the rest of the left clavicle was pulled upward. Following a standard partial (up to the second intercostal space) median sternotomy, the right internal mammary vessels were divided and ligated, and the lateral half of the sternum was transected transversely. After thoracotomy through the left second intercostal space, the second and first ribs were sectioned 10 and 3 cm from the midsternal line, respectively, and finally the left hemi-plastron was removed (Fig 3).
The tumor densely adhering to the left phrenic nerve was carefully dissected. The tumor could not be dissected from the left subclavian, internal jugular, and brachiocephalic veins. These veins were resected together with the tumor, and a reinforced ringed expanded polytetrafluoroethylene graft (Gore-Tex; W. L. Gore & Assoc, Flagstaff, AZ) was transposed between the left subclavian and brachiocephalic veins. Because the left internal jugular vein was occluded, it was simply ligated. The hemi-plastron was restored, and fixed to the sternum with stainless steel wires and to the costal cartilage with silk threads. The postoperative recovery was uneventful. The 28-month follow-up showed an asymptomatic patient with normal pulmonary function.
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Fig 3. The "hemi-plastron window" technique. (Top) The skin incision. (Bottom) The left hemi-plastron was removed and restored.
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Microscopically, there were no tumor cells at the margin of the resected specimen. The tumor seemed to arise from the left brachiocephalic vein. Hematoxylin and eosin stain showed epithelioid and spindle tumor cells with prominent intracytoplasmic vacuoles containing red blood cells, reminiscent of primitive vascular channels, in a hyaline or chondromyxoid stroma (Fig 4).
Immunohistochemical study revealed that tumor cells were positive for Factor VIII-related antigen and CD34, which are endothelial markers. The staining for epithelial membrane antigen and keratins were negative. Final pathologic diagnosis was epithelioid hemangioendothelioma.
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Fig 4. Hematoxylin and eosin stain shows epithelioid and spindle tumor cells with prominent intracytoplasmic vacuoles containing erythrocytes, in a hyaline or chondromyxoid stroma.
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Comment
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Epithelioid hemangioendothelioma is a low-grade malignant neoplasm arising from a vessel, usually a medium-sized or large vein in the liver, bone, and soft tissues [1]. Ferretti and coworkers reviewed 20 cases [2] of epithelioid hemangioendothelioma in the mediastinum. In 19 cases, except for 1 case not described in detail, the tumor arose from the anterior mediastinum, and 3 of 20 cases presented superior vena cava syndrome [2]. In spite of radiologically and pathologically aggressive appearances, long-time follow-up showed a rather indolent behavior after surgery. However, mediastinal epithelioid hemangioendothelioma has the potential to metastasize. One case was reported to present liver metastasis following mediastinal radiotherapy, and died 2 years after diagnosis [3].
Cervicothoracic tumors are usually difficult to access by classic incisions such as a median sternotomy. The hemi-clamshell incision was proposed, to provide optimal exposure of the operative field [4], which demonstrated satisfactory results from the viewpoint of postoperative morbidity and pulmonary function. Other techniques, including the transclavicular approach [5] and the transmanubrial osteomuscular sparing approach [6], were proposed. We developed a novel approach, the "hemi-plastron window" technique, which consists of (1) taking out the hemi-plastron; (2) resection of the tumor; and (3) restoring the hemi-plastron (Fig 3), which enabled us to resect the cervicothoracic tumor invading the left internal jugular, subclavian, and brachiocephalic veins with a shorter skin incision compared with the hemi-clamshell or other classic approaches.
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References
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- Weiss S.W., Enzinger F.M. Epithelioid hemangioendothelioma: a vascular tumor often mistaken for a carcinoma. Cancer 1982;50:970-981.[Medline]
- Ferretti G.R., Chiles C., Woodruff R.D., Choplin R.H. Epithelioid hemangioendothelioma of the superior vena cava: computed tomography demonstration and review of the literature. J Thorac Imaging 1998;13:45-48.[Medline]
- Begbie S.D., Bell D.R., Nevell D.F. Mediastinal epithelioid hemangioendothelioma in a patient with type IV Ehlers-Danlos syndrome: a case report and review of the literature. Am J Clin Oncol 1997;20:412-415.[Medline]
- Bains M.S., Ginsberg R.J., Jones W.G., II, et al. The clamshell incision: an improved approach to bilateral pulmonary and mediastinal tumor. Ann Thorac Surg 1994;58:30-32.[Abstract]
- Dartevelle P.G., Chapelier A.R., Macchiarini P., et al. Anterior transcervical-thoracic approach for radical resection of lung tumors invading the thoracic inlet. J Thorac Cardiovasc Surg 1993;105:1025-1034.[Abstract]
- Grunenwald D., Spaggiari L. Transmanubrial osteomuscular sparing approach for apical chest tumors. Ann Thorac Surg 1997;63:563-566.[Abstract/Free Full Text]
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S. Hasegawa, N. Isowa, and H. Wada
Reply to the editor
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Abstract
Introduction
