Ann Thorac Surg 2005;80:2351-2353
© 2005 The Society of Thoracic Surgeons
Case report
Pulmonary Sclerosing Hemangioma With Metastasis to the Mediastinal Lymph Node
Hiromichi Katakura, MD,
Manami Sato, MD,
Fumihiro Tanaka, MD,
Hiroaki Sakai, MD,
Toru Bando, MD,
Seiki Hasegawa, MD,
Yasuaki Nakashima, MD,
Hiromi Wada, MD
*
Department of Thoracic Surgery, Kyoto University, Kyoto, Japan
Accepted for publication June 16, 2004.
* Address correspondence to Dr Wada, Department of Thoracic Surgery, Kyoto University, Sakyo-ku, Shogoin, Kawahara-cho, Kyoto 606–8507, Japan (Email: wadah{at}kuhp.kyoto-u.ac.jp).
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Abstract
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During a routine health care evaluation, an abnormal shadow was detected in the chest roentgenogram of a 35-year-old man. Chest computed tomography scanning showed a nodule, approximately 3 cm in diameter, in the left S6 pulmonary segment with surrounding infiltration. Bronchoscopy revealed obstruction of the left B6c bronchus by a tumor, for which biopsy was done but no definitive histologic diagnosis could be made. Then, left lower lobectomy was performed, and the tumor was diagnosed as a pulmonary sclerosing hemangioma. A mediastinal lymph node (no. 7) showed some metastatic tumor cells. As lymph node metastasis from pulmonary sclerosing hemangioma is very rare, we herein report the details of our case.
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Introduction
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Since pulmonary sclerosing hemangioma was first reported by Liebow and Hubbell [1] in 1956, this disease has been thought to be derived from endothelial cells because of its rich vascularity. Recently, however, various diagnostic modalities, for example, immunohistochemistry and electron microscopy, have been developed, and this disease is now accepted as an epithelial neoplasm derived from the primitive respiratory epithelium, incompletely differentiated type II pneumocytes, or Clara cells [2, 3].
This disease reportedly accounts for 11% of the benign pulmonary tumors [4]. Generally, this tumor is conventionally resected without lymph node dissection because of its benign character, and tumor recurrence has been very rarely reported. Since Tanaka and associates [5] reported the first case of metastasis to the lymph nodes, 9 cases of pulmonary sclerosing hemangioma with lymph node metastasis have been reported [5–7], and our case is the 10th.
During a routine health care evaluation, an abnormal shadow was detected in the chest roentgenogram of a 35-year-old man. Chest computed tomography (CT) scanning showed a nodule, approximately 3 cm in diameter, in the left S6 pulmonary segment with surrounding infiltration (Fig 1). The blood analysis data, including tumor markers such as carcinoembryonic antigen, squamous cell carcinoma, cytokeratin 19 fragment, and pro–gastrin-releasing peptide, were within the normal range. The pulmonary function tests and arterial blood gas analysis were normal. Bronchoscopy revealed obstruction of the left B6c bronchus by a tumor for which biopsy was done, but no definitive histologic diagnosis could be made.
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Fig 1. Chest computed tomography scan showing a mass in the left S6 segment, with a relatively clear margin. Some infiltration is seen around the mass lesion.
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Because we could not rule out the possibility of malignant tumor, we resected the tumor. The tumor existed in the left S6 pulmonary segment, and its consistency was elastic hard. Intraoperative histopathological examination revealed an epithelial tumor, but we could not rule out malignancy because of artifacts in the frozen sections. Left lower lobectomy with the lymph node dissection was performed. The gross section of the resected tumor was white and sclerotic. Hematoxylin and eosin staining of paraffin-embedded sections showed a mixture of solid, sclerotic, papillary, and hemorrhagic patterns (Fig 2A). Two populations of cells could be identified; namely, solid-growing bland polygonal cells with abundant pale cytoplasm, and cuboidal lining cells covering the papillary structures (Fig 2B). The tumor cells stained positively for epithelial membrane antigen, CAM 5.2 (high molecular weight antihuman keratin), cytokeratin fragment 7, and thyroid transcription factor-1; and negatively for factor VIII and CD34 (cluster of differentiation antigen 34). These immunohistochemical results suggested that the tumor originated from the epithelium, especially from type II pneumocytes or Clara cells, but not from the endothelium. Therefore, this tumor was diagnosed as pulmonary sclerosing hemangioma. Paraffin-embedded sections of a mediastinal lymph node (no. 7) showed some metastatic tumor cells that showed the same characters as the original tumor in the lung. Because this disease has a benign nature, lymph node metastasis of pulmonary sclerosing hemangioma is very unusual.
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Fig 2. Hematoxylin-&-eosin–stained paraffin-embedded sections (x200). (A) The tumor cells extend into the normal alveolar space in solid, sclerosing, and papillary patterns. The vascular tissue is relatively dominant in the tumor. (B) The solid, sclerosing, and papillary patterns of the tumor progression are noticed in the mediastinal lymph node no. 7.
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Our patient did not receive any adjuvant therapy (neither chemotherapy nor radiotherapy) after the operation, and no recurrence has been detected at 18 months after the operation.
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Comment
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Since pulmonary sclerosing hemangioma was first reported by Liebow and Hubbell [1] in 1956, the disease has been thought to be derived from the endothelial cells because of its rich vascularity. Recently, however, various diagnostic modalities, for example, immunohistochemistry and electron microscopy, have been developed; and this disease is now accepted as an epithelial neoplasm derived from the primitive respiratory epithelium, incompletely differentiated type II pneumocytes, or Clara cells [2, 3].
Since this disease is benign pulmonary tumor, lobectomy is routinely performed and lymph node dissection is not done. In our case, as we could not confirm the diagnosis before and during operation, we dissected the hilar and mediastinal lymph nodes. Histologic examination of paraffin-embedded sections showed some metastatic cells in the no. 7 lymph node.
Nine cases of pulmonary sclerosing hemangioma with lymph node metastasis have been already reported (Table 1) [5–7], and our case is the 10th case. Of these 10 cases, the lymph node metastasis did not extend beyond the hilar nodes in 8 cases, but the mediastinal lymph nodes were involved in 2 cases including our case. The rate of lymph node metastasis is roughly estimated as 2% to 4% for this disease [5], but no cases with recurrent lymph node metastasis have been reported. Moreover, recurrence of this tumor is very rare even among the cases with lymph node metastasis. Lymph node dissection may not be necessary during surgery. Therefore, it is necessary to examine the lymph nodes during operation to determine the indication for lymph node dissection.
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References
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- Liebow AA, Hubbell DS. Sclerosing hemangioma (histiocytoma, xanthoma) of the lung Cancer 1956;9:53-75.[Medline]
- Devouassoux-Shisheboran M, Hayashi T, Linnoila RI, et al. 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]
- Chan AC, Chan JK. 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]
- Shields TW. Benign tumors of the lungIn: Malvern PA, editor. General thoracic surgery. 4th ed. New York: Williams & Wilkins; 1994. pp. 1307-1319.
- 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]
- Miyagawa-Hayashino A, Trazelaar HD, Langel DJ, et al. Pulmonary sclerosing hemangioma with lymph node metastases Arch Pathol Lab Med 2003;127:321-325.[Medline]
- Yano M, Yamakawa Y, Kiriyama M, et al. Sclerosing hemangioma with metastases to multiple nodal stations Ann Thorac Surg 2002;73:981-983.[Abstract/Free Full Text]
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Abstract
Introduction
