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Ann Thorac Surg 2002;73:981-983
© 2002 The Society of Thoracic Surgeons

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Case report

Sclerosing hemangioma with metastases to multiple nodal stations

^a Departments of Surgery II, Nagoya City University School of Medicine, Nagoya, Japan
^b Department of Radiology, Nagoya City University School of Medicine, Nagoya, Japan
^c Department of Pathology II, Nagoya City University School of Medicine, Nagoya, Japan

Accepted for publication July 16, 2001.

* Address reprint requests to Dr Yano, 1 Kawasumi, Mizuho, Nagoya, 467-8601, Japan

	Abstract

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We present a case of a large pulmonary sclerosing hemangioma with metastases to multiple lymph nodal stations and suspected contralateral pulmonary metastasis. Four cases (including the present) have been reported to have lymph node metastasis, and all had large tumors exceeding 3.5 cm in diameter. Accordingly, resection of sclerosing hemangioma is advisable while the tumor is small. Even in cases with a large sclerosing hemangioma, lymph node metastasis may be uncommon. However lymph node dissection may be necessary to detect lymph node metastasis in selected cases.

	Introduction

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Sclerosing hemangiomas (SHs) of the lung are rare, slow-growing, benign neoplasms of uncertain histogenesis. Histochemical and ultrastructural studies of this tumor have suggested epithelial (type II pneumocyte) origin [1]. However, some reports suggested mesothelial, endothelial, or neuroendocrine origins. It is also unclear as to whether SH is neoplastic. The SH was moved from the category of tumor-like lesions to that of miscellaneous tumors in the 1999 classification of the World Health Organization/International Association for the Study of Lung Cancer. A recent report of this tumor supported neoplasm theory with monoclonality using clonal analysis [2]. Thus, SH remains poorly understood. We report an unusual case of SH with metastases to multiple lymph nodal stations and suspected contralateral pulmonary metastasis.

The patient was a 67-year-old housewife with unremarkable family and medical histories. In September 1996, a routine chest roentgenogram showed an abnormal mass shadow. On the chest roentgenogram and computed tomogram a solitary, elliptic, 9 x 8 x 6–cm tumor with multiple fine calcification was pointed out (Fig 1A). It was located in the right lower lobe and adhered to the diaphragm. The enlarged subcarinal, hilar, and right inferior interlobar lymph nodes showed some calcification (Fig 1B). The pathologic samples of percutaneous biopsy indicated sclerosing hemangioma. In February 1997, right lower lobectomy was performed. The tumor showed tight adhesion to the diaphragm. A part of the diaphragm was resected with the tumor. Additionally, hilar and mediastinal lymph nodes were resected. In the pathologic findings of the tumor, a sclerotic component with dystrophic calcification was predominant, with minor papillary or solid components with hemorrhage. The tumor had no capsule and extended directly into the intact lung. The diaphragm was pathologically intact without tumor invasion. The lymph nodes (3, 7, 9, 11s, and 11i) had metastatic lesions with sclerosis and calcification. Eleven months after the first operation, on the follow-up chest roentgenogram and computed tomogram, a small coin lesion was pointed out in the ventral segment of the left upper lobe. Partial resection of the left upper lobe was performed under video-assisted thoracoscopy. The pathologic findings showed a typical finding of sclerosing hemangioma with papillary and hemorrhagic components but with less severe sclerosis compared with the first tumor. After another 3 years of observation, no recurrent or metastatic findings have been found.

View larger version (83K): [in this window] [in a new window]   Fig 1. Chest radiograph computed tomographic scan demonstrating a right lower lobe mass with multiple fine calcification adhered to the diaphragm (A). Subcarinal and hilar lymph node enlargement was noted, with multiple calcifications (B).

	Comment

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In typical cases, four major pathologic patterns are identified (solid, hemorrhagic, papillary, and sclerotic components), and one component tends to predominate. In our case, the sclerotic component with calcification was obviously predominant in the lung and in the lymph nodes. Also in the lymph node lesions, sclerosis with calcification becomes further dominant. Surface cuboidal cells in papillary structures of both the primary tumor and the lymph node lesions were positively immunostained for surfactant apoprotein (Fig 2A). Not only surface cuboidal cells but also round pale cells were stained for epithelial membrane antigen (Fig 2B). These findings were consistent with a type II pneumocyte origin [1]. We considered the present case to be a case of SH of the lung with lymph node metastasis.

View larger version (124K): [in this window] [in a new window]   Fig 2. Immunostains of the main tumor. (A) Cuboidal cells in papillary structures are positive for surfactant apoprotein. (B) Not only surface cuboidal cells but also round pale cells were stained for epithelial membrane antigen. (Surfactant apoprotein staining (A); epithelial membrane antigen staining (B); x200.)

In 1986, Tanaka and colleagues [4] reported the first case of SH with lymph node metastasis. In only one of the dissected hilar lymph nodes was a small metastatic deposit found. Devouassoux-Shisheboran [5] and associates reported a series of 100 SH cases. This series included a case with peribronchial lymph node metastasis. Chan and Chan [6] reported a case of SH with hilar lymph node metastasis. These three cases showed single lymph node metastasis that was found incidentally as microscopic disease. The present case was an unusual case that was clinically diagnosed with metastases to multiple lymph nodal stations. Similar to our case, in all reported cases of SH with concurrent node metastasis, the size of the primary tumor was great as 5 cm [4], 3.5 [5], and 8 cm [6]. Conversely, in the series of Devouassoux-Shisheboran and associates [5], 73.7% of SH showed a diameter of less than 3 cm. As a consequence, lymph node metastasis may be a characteristic of a large SH although lymph node metastasis may occur uncommonly. Accordingly, resection of SH is advisable while the tumor is small. Lymph node dissection may be necessary to detect lymph node metastasis in cases with a large SH or with enlarged lymph nodes that may be due to metastasis.

	References

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1. Nagata N., Dairaku M., Sueishi K., Tanaka K. Sclerosing hemangioma of the lung. An epithelial tumor composed of immunohistochemically heterogenous cells. Am J Clin Pathol 1987;88:552-559.[Medline]
2. Niho S., Suzuki K., Yokose T., Kodama T., Nishiwaki Y., Esumi H. Monoclonality of both pale cells and cuboidal cells of sclerosing hemangioma. Am J Pathol 1998;152:1065-1069.[Abstract]
3. Liebow A.A., Hubbell D.S. Sclerosing hemangioma (histiocytoma) of the lung. Cancer 1956;9:53-75.[Medline]
4. 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]
5. 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]
6. Chan A.C.L., Chan J.K.C. Pulmonary sclerosing hemangioma consistently expresses thyroid transcription factor-1 (TTF-1). A new clue to its histogenesis. Am J Surg Pathol 2000;24:1531-1536.[Medline]

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