Sclerosing hemangioma.

To the Editor: We recently evaluated a 48-year-old, nonsmoking Filipino female with a 3-week history of cough and hemoptysis. Physical examination was unremarkable. Chest radiography and computed tomography scan of the chest showed a 6.0-cm mass in the right lower lobe. A purified protein derivative skin test was positive, and three sputum samples were obtained. The patient underwent a transthoracic needle aspiration of the right lower lobe mass as well as fiberoptic bronchoscopy. All studies were nondiagnostic.

The patient subsequently underwent a thoracotomy with right lower lobectomy. Microscopic sections of the subpleural mass showed several distinct histologic patterns, including (1) solid sheets of round cells, (2) areas of sclerosis, (3) papillary structures with "cores" of round cells and a lining of squamous-to-cuboidal cells, and (4) hemorrhagic cysts with the same type of lining as the papillary structures. These findings were consistent with a lesion known as sclerosing hemangioma (SH). In addition, cut sections of the surrounding lung showed scattered necrotizing granulomata. Although mycobacterial and fungal stains were negative, empiric treatment for tuberculosis was initiated, pending culture results. Mycobacterium tuberculosis was eventually confirmed on culture, and the patient successfully completed a 6-month course of treatment.

SH of the lung is a rare, benign pulmonary neoplasm first described by Liebow and Hubbell in 1956. (1) Small case series indicate that patients range in age from 13 to 76 years (mean, 46) and that there is a female preponderance of approximately 5:1. (2,3) SH has been reported in several ethnic groups including whites, blacks, Asians, and Hispanics. (2) The majority of lesions are noted incidentally on routine chest imaging. If symptoms are present, they may include hemoptysis, cough, and chest pain, but the majority of patients are asymptomatic. (2)

The typical chest radiograph shows a solitary, well-circumscribed, intraparenchymal lesion ranging from 0.3 to 8.2 cm (mean, 3.6 cm). (2,3) Less common findings of SH include multifocal lesions, hilar lymph node metastasis, endobronchial disease, pleural disease, and mediastinal disease. (2,3) All cases of metastatic disease have occurred in the context of a large primary tumor (diameter >3.5 cm).

Diagnosis of SH generally requires an open lung biopsy. With limited sampling techniques, SH may be confused with well-differentiated adenocarcinoma or carcinoid tumor. (4) Definitive diagnosis may ultimately rest on analysis of multiple routine microscopic sections of a resected specimen, with additional immunohistochemical and ultrastructural studies as needed. (2) In all cases, SH contain two or more of the following four major histologic patterns: solid, hemorrhagic, papillary, and sclerotic.

Long-term follow-up data is limited; however, Katzenstein et al (3) followed 22 cases of completely resected patients for an average of 3.6 years without evidence of recurrence. Yano et al (5) reported a contralateral coin lesion that appeared 11 months after the resection of SH. The authors could not conclude whether this represented a pulmonary metastasis or multiple heterochronous tumors.

In summary, SH is an uncommon pulmonary lesion that occurs in middle-aged female patients. Most cases are asymptomatic, and the lesion is incidental. Pulmonary or systemic symptoms should prompt a search for concurrent disease. In our patient, this disease was pulmonary tuberculosis.

References

1. Liebow A, Hubbell D. Sclerosing hemangioma (histiocytoma, xanthoma) of the lung. Cancer 1956;9:953-975.

2. Devouassoux-Shisheboran M, Hayashi T, Linnoila R, et al. A clinicopathologic study of 100 cases of pulmonary sclerosing hemangioma with immunohistochemical studies. Am J Surg Pathol 2000;24:906-916.

3. Katzenstein A, Gmelich J, Carrington C. Sclerosing hemangioma of the lung: a clinicopathologic study of 51 cases. Am J Surg Pathol 1980;4:343-356.

4. Ng W, Fu K, Wang E, et al. Sclerosing hemangioma of the lung: a close cytologic mimicker of pulmonary adenocarcinoma. Diagn Cytopathol 2001;25:316-320.

5. Yano M, Yamakawa Y, Kiriyama M, et al. Sclerosing hemangioma with metastases to multiple nodal stations. Ann Thorac Surg 2002;73:981-983.

Christopher M. Stafford, MD

Stephen W. Crawford, MD, FCCP

David A. Bradshaw, MD

Guerard P. Grice, MD

Divisions of Pulmonary Medicine and Pathology

Naval Medical Center San Diego

San Diego, CA