© 2005 Foundation for Promotion of Cancer Research
Case Report |
Multiple Sclerosing Hemangiomas with a 10-year History
1 Divison of Thoracic Oncology, National Cancer Center Hospital East, Kashiwa, Chiba and 2 Pathology Division, National Cancer Center Research Institute East, Kashiwa, Chiba, Japan
For reprints and all correspondence: Kanji Nagai, Division of Thoracic Oncology, National Cancer Center Hospital East, 6-5-1, Kashiwanoha, Kashiwa, Chiba 277-8577, Japan. E-mail: knagai{at}east.ncc.go.jp
Received July 6, 2004; accepted August 22, 2004
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Abstract |
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We report a case of multiple sclerosing hemangiomas arising in a 38-year-old woman. Computed tomography (CT) scans of the chest showed multiple small nodules in all lobes of the right lung. The nodule sizes ranged from a few millimeters to the largest of 3.1 cm, which was located in the right middle lobe. She underwent right middle lobectomy to confirm the histological diagnosis. The resected specimen revealed multiple sclerosing hemangiomas. We followed this patient by annual chest CT scans for 10 years and demonstrated that all residual nodules remained unchanged. This is the first report of stable multiple pulmonary sclerosing hemangiomas observed for such a long period.
Key Words: sclerosing hemangioma • multiple • lung
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INTRODUCTION |
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TOPAbstractINTRODUCTIONCASE REPORTDISCUSSIONReferences |
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Pulmonary sclerosing hemangioma is a rare lung neoplasm usually presenting as a peripheral and solitary nodule of less than 3 cm in diameter, predominantly in asymptomatic middle-aged women (1,2). The clinical course of sclerosing hemangioma is usually slow and benign. However, as they are extremely rare manifestations, their natural course has not been well understood.
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CASE REPORT |
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TOPAbstractINTRODUCTIONCASE REPORTDISCUSSIONReferences |
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A 38-year-old asymptomatic woman underwent chest roentography for preoperative evaluation of bartholinitis. Multiple small nodules were pointed out in the right lung field, and she was referred to our institution for further examinations. Computed tomography (CT) scans of the chest showed innumerable lesions varying from several millimeters to 3.1 cm in diameter in all lobes of the right lung but none in the left lung. Each lesion was a round-shaped nodule with a distinct margin and homogeneous density. The largest lesion, measuring 3.1 cm x 2.9 cm, was depicted in the right middle lobe accompanied by numerous tiny nodules (Fig. 1). In the right upper and lower lobes, multiple nodules of various sizes were present. No lymphadenopathy was observed. Bronchoscopic examinations failed to obtain sufficient material because of massive bleeding from the largest middle lobe lesion. She underwent right middle lobectomy in September 1993 in order to confirm the histological diagnosis. She was discharged without any complications on the seventh postoperative day.
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Macroscopically, a well-circumscribed nodule measuring 2.7 cm x 2.7 cm in size was located in the right middle lobe of the resected specimen. Scattered around the main nodule were observed innumerable minute nodules of various sizes. They were well-circumscribed but not encapsulated. Microscopically, each nodule was composed of a variable proportion of four patterns: solid, hemorrhagic, sclerotic and papillary. Smaller lesions, particularly minute ones, were mainly of solid and hemorrhagic patterns. The solid pattern was composed of a sheet-like proliferation of round to polygonal cells with pale cytoplasm. In the hemorrhagic pattern, red blood cells were accumulated within cystic spaces composed of cuboidal surface cells (Fig. 2). In a small portion of the tumor, the papillary pattern composed of cuboidal surface cells was observed within the solid proliferation of round tumor cells (Fig. 3). The sclerotic pattern was scarcely present. Based on these histological findings, we diagnosed these lesions as multiple sclerosing hemangiomas of the lung.
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Postoperatively, the patient visited our outpatient clinic at 6-month intervals for 3 years and at 1-year intervals thereafter for 7 years. We have been following up residual lesions located in the right upper and lower lobes by chest roentography at every visit and annual chest CT scans. Figure 4a and b shows the comparative CT scans of the right lower lobe lesions taken in March 1995 (about 18 months after surgery) and in March 2004 (about 10 and a half years after surgery). There was neither nodule growth nor new lesions.
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DISCUSSION |
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TOPAbstractINTRODUCTIONCASE REPORTDISCUSSIONReferences |
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Sclerosing hemangioma of the lung is a relatively rare benign tumor first described by Liebow and Hubell in 1956 (1). Histologically, there is a mixture of solid, sclerotic, papillary and hemorrhagic components in typical cases. In each component, two populations of cells can be identified: solid-growing polygonal round cells with pale cytoplasm and cuboidal surface cells covering papillary structures (2). Recent studies suggest that sclerosing hemangioma derives from primitive respiratory epithelium and demonstrates neoplastic differentiation (3,4). The majority of pulmonary sclerosing hemangiomas present as a solitary pulmonary nodule, whereas multiple lesions are reported to account for 4% of all cases (2,3). Because of its rarity, the natural course of pulmonary multiple sclerosing hemangiomas has not been well understood.
CT findings of residual nodules in the present case were very similar to resected nodules, and the resected middle lobe contained numerous foci of sclerosing hemangiomas. Therefore, we believe residual scattered nodules are multiple sclerosing hemangiomas, although cytopathological evidence was not obtained. It has not been fully studied whether the multiplicity of multiple sclerosing hemangiomas suggests a progressive clinical course or not.
As we reviewed the literature, although a solitary pulmonary sclerosing hemangioma that showed rapid progression was reported (5), multiple lesions previously reported have all been slow-growing cases. Hayashi and colleagues followed a patient with gradually growing multiple pulmonary sclerosing hemangiomas for as long as 7 years before surgical resection (6). Lee and coworkers followed a case of bilateral multiple (four) sclerosing hemangiomas of the lung, which grew slowly (7). We have followed the present case intensively using CT scans for 10 years and demonstrated no changes in residual lesions. These cases suggest that multiplicity of pulmonary sclerosing hemangioma does not necessarily imply biological aggressiveness. Biological behavior might be different between solitary and multiple cases. However, further analysis of a larger group of patients is required.
In conclusion, we reported a rare case of multiple sclerosing hemangiomas of the lung unchanged for over 10 years. This case and review of the literature suggest that the multiplicity of multiple sclerosing hemangiomas does not necessarily indicate a dismal outcome.
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Acknowledgments |
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We thank Professor J. Patrick Barron, Tokyo Medical University, for reviewing the English manuscript. This work was supported in part by a Grant-in-Aid for Cancer Research from the Ministry of Health, Labour and Welfare in Japan.
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References |
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TOPAbstractINTRODUCTIONCASE REPORTDISCUSSIONReferences |
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1 Liebow AA, Hubbell DS. Sclerosing hemangioma (histiocytoma, xanthoma) of the lung. Cancer 1956;9:53–75.[CrossRef][Medline]
2 Colby TV, Koss MN, Travis WD. Tumors of the lower respiratory system. In Rosai J, Rosai JS, editors, Atlas of tumor pathology, 3rd series, Fascicle 13. Washington, DC: Armed Forces Institute of Pathology 1995; 465–71.
3 Devouassoux-Shisheboran M, Hayashi T, Linnoila RI, Koss MN, Travis WD. 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;24:906–16.[CrossRef][ISI][Medline]
4 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;152:1065–9.[Abstract]
5 Shibata R, Mukai M, Okada Y, Sakamoto M, Yamauchi T, Kobayashi K. A case of sclerosing hemangioma of the lung presenting as a gigantic tumor occupying the left thoracic cavity. Virchows Arch 2003;442:409–11.[Medline]
6 Hayashi A, Takamori S, Mitsuoka M, et al. Unilateral progressive multiple sclerosing hemangioma in a young female successfully treated by pneumonectomy: report of a case. Int Surg 2002;87:69–72.[Medline]
7 Lee ST, Lee YC, Hsu CY, Lin CC. Bilateral multiple sclerosing hemangiomas of the lung. Chest 1992;101:572–3.
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