Ann Thorac Surg 2003;76:1275-1276
© 2003 The Society of Thoracic Surgeons
Case report
A pulmonary cavernous hemangioma causing massive hemoptysis
Mehmet S
rmal, MDa*,
Funda Demira
, MDb,
Ertan Aydn, MDa,
Sezgin Karasu, MDa,
Sadi Kaya, MDa
a Department of Thoracic Surgery, Atatürk Training and Research Hospital for Chest Disease and Chest Surgery, Ankara, Turkey
b Department of Pathology, Atatürk Training and Research Hospital for Chest Disease and Chest Surgery, Ankara, Turkey
Accepted for publication November 27, 2002.
* Address reprint requests to Dr Srmal, Kardelen mahallesi Mobilyaclar sitesi, 482/19 Batkent, Ankara, Turkey
e-mail: mehmetsirmali{at}yahoo.com
 |
Abstract
|
|---|
A 54-year-old man was seen with massive hemoptysis. A posteroanterior chest radiograph revealed increased bronchovascular branching in the left hemithorax. Thoracic computed tomography showed a well-demarcated mass with uniform density confined to the apicoposterior and anterior segments of the left upper lobe. Fiberoptic bronchoscopy revealed a locus of bleeding in the apicoposterior segment of that lobe. Exploratory left thoracotomy confirmed the presence of a mass. The results of frozen section examination of a biopsy specimen were benign. The mass was resected by upper lobectomy. The definitive result of histopathological study of the mass was cavernous hemangioma. Very few cases of pulmonary hemangioma have been reported in the literature.
|
Introduction
|
|---|
Hemangiomas comprise closely situated tangles of capillaries or widely dilated vascular channels that have focal connective tissue in their walls and that are coated with squamous endothelial cells. They are frequently located in the liver, the skin, and the subcutaneous tissues [1]. Although they can affect any organ, very rarely do hemangiomas occur as a primary tumor of the lungs [2].
A 54-year-old man sought medical attention because of massive hemoptysis. He had experienced two bouts of hemoptysis; the first had occurred some 10 days previously and the second, 3 days later. The patient reported the loss of approximately 500 mL of blood each time. After the last major bout, he had hemoptysis while coughing eight to ten times a day, especially in the mornings. The history was unremarkable, and the findings on physical examination were normal. A posteroanterior chest radiograph revealed prominence of the interstitial markings in the left hemithorax. Thoracic computed tomographic scans showed a well-defined solid mass 4 x 3 cm in the apicoposterior and anterior segments of the left upper lobe (Fig 1).
The mass was in contact with the mediastinal pleura and continuous with the left upper vascular bundle. No prominent infiltration was observed at the periphery of the lesion.
View larger version (89K):
[in this window]
[in a new window]
|
Fig 1. Computed tomographic scans (A: mediastinal window), (B: parenchymal window) showing a well-demarcated solid mass continuous with the vascular bundle in the anterior segment of the left upper lobe.
|
|
The patient underwent emergency fiberoptic bronchoscopy. Mucosal irregularities were present at the site where the left main bronchus emerged. An active locus of bleeding in the apicoposterior segment was noted. The findings from cytological study of bronchial lavage were benign. Next day exploratory left thoracotomy was performed. A smooth, medium-hard mass measuring 5 x 4 cm was identified in the apicoposterior and anterior segments. There was a minute space between the perimeter of the mass and the superior pulmonary vein. A biopsy sample was taken by wedge resection and sent for frozen-section examination. There was excessive bleeding after the biopsy. The results of the frozen-section study were benign.
Wedge resection of the mass was not possible because of its close proximity to the superior pulmonary vein; instead, upper lobectomy was performed. Postoperative histopathological study identified the mass as a pulmonary cavernous hemangioma. Hemoptysis disappeared in the postoperative period. To exclude the possibility of an abdominal hemangioma, the patient underwent abdominal ultrasound; the results were normal.
The patient was discharged on the ninth postoperative day. In the monthly follow-up visit, he was healthy both clinically and radiologically.
|
Comment
|
|---|
Cavernous hemangiomas, a variant of pulmonary arteriovenous malformations, rarely occur in the lung [2, 3]. The age of such patients in the literature ranges between 10 weeks and 61 years. Hemangiomas affect both sexes equally. When hemangioma occurs in the lung, it can present as a solitary mass or multiple masses [4–6]. In the case of our patient, there was a single mass confined to the apicoposterior segment of the upper lobe of the left lung.
Histopathologically hemangiomas are composed of tangles of capillaries or widely dilated vascular channels. Their walls contain focal connective tissue and are coated with squamous endothelial cells [1]. Microscopic examination of the specimen from our patient revealed a nonencapsulated but well-circumscribed nodule comprising big, dilated vessels lined with squamous endothelium and filled with blood. The vessels were distributed diffusely between the bronchioles and the alveoli. The walls of the vessels were thickened with adventitial fibrosis (Fig 2).
Computed tomography and magnetic resonance imaging of the thorax have been useful for preoperative diagnosis [2, 6]. However, the definitive diagnosis can be made by histopathological examination.
Symptoms related to cavernous hemangioma depend on the location and the size of the lesion and the number of lesions. Symptoms include respiratory distress, cyanosis, and hemoptysis, though asymptomatic cases have been reported [2–6]. Our patient was seen with massive hemoptysis.
Solitary cavernous hemangioma should be treated with surgical excision [2]. We surgically excised the mass in our patient. However, Wu and colleagues [4] reported that they used interferon-alfa-2a successfully to treat a 7-year-old patient with pulmonary cavernous hemangioma with respiratory distress and hemoptysis.
When cavernous hemangioma is suspected, it is necessary to determine whether the lesion is associated with the endothelium. This can be achieved by immunohistological staining of the cells lining the cavernous lumen. Cavernous hemangiomas show positive staining for anti–von Willebrand factor antibody and negative staining for anti–epithelial membrane antibody [6]. In our patient the endothelium showed a strong positivity to CD31 and CD34, immunohistochemical markers of vessels.
Cavernous hemangiomas are rare lesions of the lungs. They can be asymptomatic or can present with life-threatening symptoms such as massive hemoptysis. The choice of treatment of pulmonary cavernous hemangioma is surgical resection.
|
References
|
|---|
- Cotran R.S., Kumar V., Collins T., Robbins S.L. Pathologic basis of disease. Philadelphia: WB Saunders, 1999.
- Shields T.W., Robinson P.G. Benign tumors of the lung. In: Shields T.W., LoCicero J., Ponn R.B., eds. General thoracic surgery. Philadelphia: Lippincott Williams & Wilkins, 2000:1523.
- Galliani C.A., Beatty J.F., Grosfeld J.L. Cavernous hemangioma of the lung in an infant. Pediatr Pathol 1992;12:105-111.[Medline]
- Wu J.M., Lin C.S., Wang J.N., Luo C.Y., Yu C.Y., Yang H.B. Pulmonary cavernous hemangiomatosis treated with interferon alfa-2a. Pediatr Cardiol 1996;17:332-334.[Medline]
- Mori A., Obata K., Tada T., Yagawa K., Satoh M. A case of multiple cavernous hemangiomas of the lung. Gan No Rinsho 1985;31:1433-1438.[Medline]
- Kase M., Sakamoto K., Yamagata T., et al. A case of pulmonary cavernous hemangioma: immunohistological examination revealed its endothelial cell origin. Kyobu Geka 2000;53:1055-1057.[Medline]
Top
Abstract
Introduction
