Ann Thorac Surg 2003;75:1021-1023
© 2003 The Society of Thoracic Surgeons
Case report
Sclerosing hemangioma isolated to the mediastinum
Kazuhiro Sakamoto, MD*a,
Masato Okita, MDa,
Hiroshi Kumagiri, MDa,
Shunji Kawamura, MDa,
Koichiro Takeuchi, MDa,
Riichiro Mikami, MDa
a Department of Respiratory Surgery, Yokohama Rosai Hospital, Yokohama, Japan
Accepted for publication August 24, 2002.
* Address reprint requests to Dr Sakamoto, First Department of Surgery, Yokohama City University School of Medicine, 3-9 Fukuura, Kanazawa-ku, Yokohama 236-0004, Japan.
e-mail: saka784{at}lycos.jp
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Abstract
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Sclerosing hemangioma is an uncommon tumor of unknown histogenesis that generally develops in the lung. We report on a 48-year-old woman with a sclerosing hemangioma that was apparently isolated to the mediastinum. To our knowledge, sclerosing hemangioma arising in the mediastinum has not been previously reported. Potential mechanisms explaining the isolation of sclerosing hemangioma in the mediastinum are discussed.
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Introduction
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Sclerosing hemangioma (SH) of the lung is a rare tumor whose histogenesis is poorly understood. It usually arises in the periphery of the lung as a solitary tumor measuring less than 5-cm diameter [1]. SH of the mediastinum is extremely rare. One case of mediastinal lymph node metastasis from SH of the lung has been reported [2]. We describe a case of SH that apparently isolated to the mediastinum, with no evidence of SH in the lung.
A healthy 48-year-old Japanese woman was admitted for an abnormal shadow on a chest roentgenogram film. Laboratory data were normal, including normal carcinoembryonic antigen, squamous cell carcinoma antigen, and neuron specific enolase. A computed tomographic scan reveald a 7 x 5 cm mass in the posterior mediastinum (Fig 1). Transesophageal ultrasonography illustrated that the mass contained multiple cystic cavities. Magnetic resonance imaging of the chest revealed that the mass in the mediastinum compressed the adjacent left atrium and the right bronchus (Fig 2).
Fiberoptic bronchoscopy demonstrated no abnormality, except for external compression of the right bronchus. A provisional diagnosis of bronchogenic cyst was made, and the patient underwent an operation through a right posterolateral thoracotomy. There was no palpable lung mass. The tumor was located in the posterior mediastinum and was in close contact with the pericardium. It was covered by the mediastinal pleura and demonstrated no apparent connection with the lung. Therefore, we could bluntly dissect and isolate the tumor. There was no evidence that the tumor had originated in the lung and invaded the mediastinum (i.e., the tumor appeared to have isolated to the mediastinum).
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Fig 2. A coronal T1-weighted magnetic resonance image (TR/TE = 909/11) illustrating the tumor below the carina. The tumor compresses the right bronchus.
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On gross examination, the tumor measured 7 x 5 x 4.5 cm and was dark red and well circumscribed, but not encapsulated. The cut surface of the tumor consisted of multiple blood-filled cysts with solid components. On microscopic examination, the tumor exhibited four histologic patterns: solid, papillary, sclerotic, and hemorrhagic. The sclerotic area contained calcifications. The tumor cells were interstitial epithelioid cells. Hyperplastic type II pneumocytes lined the surface of the papillary and cystic structures (Fig 3).
Mitotic figures were rare in all sections examined. The interstitial epithelioid cells were immunoreactive with epithelial membrane antigen, vimentin, chromogranin, and thyroid transcription factor-1 (TTF-1) and were negative for Factor VIII-related antigen, CD34, and synaptophysin immunostains. There was no evidence of visceral pleura or lung parenchyma. Based on these results, the tumor was diagnosed as a SH isolated to the mediastinum.
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Fig 3. Microscopic features of the resected specimen. Solid and hemorrhagic patterns are observed. Sheets of round tumor cells with eosinophilic or clear cytoplasm are present in the solid structures. Hyperplastic type II pneumocytes line the surface of the cystic structures (hematoxylin and eosin stain, magnification x100).
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The patient’s postoperative recovery was uneventful. Five-years postoperatively, the patient was well with no evidence of recurrence.
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Comment
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Liebow and Hubbell [3], the first to describe SH, suggested that this benign tumor was an endothelial proliferation; however, electron microscopic and immunohistochemical studies suggest that SH originates from epithelial cells [4, 5]. The exact histogenesis of SH thus remains uncertain [1, 5].
This is the first report to describe SH isolated to the mediastinum. One case of mediastinal SH was found among a series of 100 patients with pulmonary SH, but was considered to have arisen in the periphery of the lung and protrude into the mediastinum, mimicking mediastinal SH [6]. Many investigators consider that SH develops from type II alveolar epithelial cells, although this remains controversial. Positive TTF-1 staining supports the hypothesis that the tumor originated from respiratory epithelial cells [6]. In our patient, however, the tumor developed outside the lung.
Three mechanisms may account for the development of mediastinal SH. The first is metastasis from the lung to the mediastinum, although there was no evidence of tumor in the lung. All previously reported cases of lymph node metastasis from SH had evidence of primary tumor measuring at least 3.5-cm diameter in the lung [2, 6, 7]. The second mechanism is that the tumor could have originated from ectopic lung tissue, such as bronchogenic cysts. However, no ectopic lung tissue was found in the surgical specimen. The final mechanism is that the SH developed from the lung as a pedunculated pleural mass that slowly pulled away from the lung surface.
All three of these potential mechanisms are plausible; however, none can satisfactorily explain the apparent extrapulmonary location of the SH in our patient. Although we consider this to be the first documented case of SH isolated to the mediastinum, immunohistochemical studies suggest that the tumor arose from lung tissue. Our report demonstrates that, although rare, SH should be considered in the differential diagnosis of mediastinal masses.
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Acknowledgments
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We thank Yukio Nakatani, MD, Tamiko Takemura, MD, Tomayoshi Hayashi, MD, and William D. Travis, MD, for pathologic support.
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References
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- Sugio K., Yokoyama H., Kaneko S., Ishida T., Sugimachi K. Sclerosing hemangioma of the lung: radiographic and pathological study. Ann Thorac Surg 1992;53:295-300.[Abstract]
- Yano M., Yamakawa Y., Kiriyama M., Hara M., Murase T. Sclerosing hemangioma with metastases to multiple nodal stations. Ann Thorac Surg 2002;73:981-983.[Abstract/Free Full Text]
- Liebow A.A., Hubbell D.S. Sclerosing hemangioma (histiocytoma, xanthoma) of the lung. Cancer 1956;9:53-57.[Medline]
- Hill G., Eggleston J. Electron microscopic study of so-called "sclerosing hemangioma": report of a case suggesting epithelial origin. Cancer 1972;30:1092-1106.[Medline]
- Rodriguez-Soto J., Colby T.V., Rouse R.V. A critical examination of the immnophenotype of pulmonary sclerosing hemangioma. Am J Surg Pathol 2000;24:442-450.[Medline]
- Devouassoux-Shisheboran M., Hayashi T., Linnoila R.I., Koss M.N., Travis W.D. A clinicopathologic study of 100 cases of pulmonary sclerosing hemangioma with immunohistochemical studies: TTF-1 is expressed in both round and surface cells, suggesting an origin from primitive respiratory epithelium. Am J Surg Pathol 2000;24:906-916.[Medline]
- Tanaka I., Inoue M., Matsui Y., et al. A case of pneumocytoma (so-called sclerosing hemangioma) with lymph node metastasis. Jpn J Clin Oncol 1986;16:77-86.[Abstract/Free Full Text]
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Abstract
Introduction
