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J Thorac Cardiovasc Surg 2004;127:1843-1845
© 2004 The American Association for Thoracic Surgery
Brief communication |
a Department of Cardiovascular Surgery, Nagoya National Hospital, Nagoya, Japan,
b Department of Pathology, Nagoya National Hospital, Nagoya, Japan
c Department of Cardiovascular Surgery, Nagoya University Graduate School of Medicine, Nagoya, Japan
Received for publication November 1, 2003; revisions received December 24, 2003; accepted for publication January 8, 2004.
* Address for reprints: Masaho Okada, MD, Department of Cardiovascular Surgery, Nagoya University School of Medicine, 65 tsurumaicho, Shouwa-ku, Nagoya, Aichi 466-8560, Japan
OKMASAHO{at}aol.com
Clinical summary
A 50-year-old man who was given a diagnosis of Marfan syndrome was hospitalized because of a right visual field defect. He had undergone a Cabrol operation for annuloaortic ectasia and aortic valve regurgitation 17 years earlier. His ascending aorta and aortic root were replaced with a woven Dacron graft (DeBakey Vasculour; C.R. Bard, Inc, Murray Hill, NJ) and a mechanical valve (Björk-Shiley monostrut heart valve, 25 mm; Shiley, Inc, Irvine, Calif). He underwent mitral valve replacement with a mechanical valve for mitral valve insufficiency caused by acute endocarditis 5 years earlier. He was hospitalized because of right-side hemiparesis but fell into a semicoma thereafter. Computed tomography (CT) revealed a left occipital brain tumorous shadow. A tumorectomy of the left occipital lobe was performed, but the patient died as a result of an expanding brain hemorrhage 2 weeks later. The brain tumor was diagnosed as metastatic angiosarcoma.
Autopsy findings
The aortic root had a wrap inclusion of the aneurysmal sac surrounding a woven Dacron aortic graft. There was a tumorous mass along the noncoronary cusp of the anastomosed site, with soft granulation tissue in it (Figure 1). The tumor extended into the aneurysmal sac and invaded the graft inside as well. The brain had multiple metastatic tumors and expanding hemorrhaging. There were no metastatic tumors in the other organs.
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Discussion
Foreign bodies have been shown to induce sarcomas in experimental animals,1 but it has only rarely been reported in human subjects that sarcomas have developed adjacent to foreign material introduced into the body either iatrogenically or accidentally.2 In thoracic surgery various forms of Dacron prostheses are commonly used. Oppenheimer and colleagues1 briefly mentioned that the common denomination of tumor-producing materials was a long-chain polymer structure, such as Dacron. Seven sarcomas have been reported arising in association with Dacron vascular prostheses in the English-language literature.3-6 The present case is angiosarcoma arising from the anastomosed site of the aortic root with a Dacron aortic graft. Neoplastic cells strongly expressed CD31, a reliable marker that has both relative specificity and excellent sensitivity to angiosarcomas of all types,7,8 confirming the pathologic diagnosis. Neoplastic cells infiltrated both sides of the endothelial layer of the graft from the anastomosed site. Therefore we speculate that the anastomosed site with a Dacron graft is the origin of the angiosarcoma. Angiosarcoma has also been reported to have developed at the site of defunctionalized arteriovenous fistulas.9,10 The wrap inclusion of the aneurysmal sac around the aortic root is one example of a defunctionalized arteriovenous fistula. There was a thrombus with neoplastic cells in it. An exuberant host response around the foreign material might represent an important intermediate step in the development of the sarcoma.11 The cells in the inflammatory or repair process rarely undergo a malignant transformation, which is probably associated with an oncogene activation and a tumor suppressor gene inactivation.3 Sarcomas associated with foreign material commonly have a long latent period of more than a decade.
Primary tumors of the aorta are rare, and the diagnosis is difficult. Most of the reported cases were diagnosed after operation or autopsy. The present case was also diagnosed first to be angiosarcoma with specimens of brain tumors. CT scanning supplemented with angiography helps to delineate masses ,and magnetic resonance imaging can define the extent of the invasion and might allow the histogenetic classification.12
The presented patient had a previous CT tomography 5 years ago. There was an expansion of the low-density area at the aortic root surrounding the graft compared with the recent scan retrospectively. Hematoma or tumorous mass had expanded in the aneurysmal sac around the aortic root for the last 5 years.
The prognosis of angiosarcoma depends on the site of origin and on the tumor size but generally is grave.13 The origin of the angiosarcoma could not be diagnosed antemortem, but this is the first cardiac angiosarcoma in association with a Dacron graft in the aortic root.
References
This article has been cited by other articles:
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  N. D. Saunders, J. S. Marshall, and R. C. Anderson A case of chest wall angiosarcoma associated with breast implants. J. Thorac. Cardiovasc. Surg., October 1, 2007; 134(4): 1076 - 1077. [Full Text] [PDF]
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