CASE REPORT

The Clinical Respiratory Journal

Venous hemangioma of the anterior mediastinum Adnan Yilmaz1, Murat Kavas1, Ays¸e Ersev2, Gulbanu Horzum Ekinci1, Osman Haciomeroglu1, Cansel Atinkaya3 and Esen Akkaya1 1 Pulmonology, Süreyyapas¸a Center for Chest Diseases and Thoracic Surgery Training and Investigation Hospital, Istanbul, Turkey 2 Pathology, Süreyyapas¸a Center for Chest Diseases and Thoracic Surgery Training and Investigation Hospital, Istanbul, Turkey 3 Thoracic Surgery, Süreyyapas¸a Center for Chest Diseases and Thoracic Surgery Training and Investigation Hospital, Istanbul, Turkey

Abstract Mediastinal hemangiomas are rare tumors, with an incidence of 0.5% or less. We herein present a case of venous hemangioma in the anterior mediastinum. A 61-year-old man was admitted to our hospital complaining of dyspnea. Computed tomography scan of the thorax showed a 2.5 × 3.2 × 2 cm mass in the anterior mediastinum. Using a median sternotomy approach, the tumor was completely removed. Pathological examination confirmed a venous hemangioma. Please cite this paper as: Yilmaz A, Kavas M, Ersev A, Ekinci GH, Haciomeroglu O, Atinkaya C and Akkaya E. Venous hemangioma of the anterior mediastinum. Clin Respir J 2014; ••: ••–••. DOI:10.1111/crj.12241.

Key words anterior mediastinum – sternotomy – vascular tumor – venous hemangioma Correspondence Adnan Yilmaz, MD, Pulmonology, Süreyyapas¸a Center for Chest Diseases and Thoracic Surgery Training and Investigation Hospital, 34843 Istanbul, Turkey. Tel: +905322877497 Fax: +902164214130 email: [email protected] Received: 27 July 2014 Accepted: 27 October 2014 DOI:10.1111/crj.12241 Authorship and contributorship Adnan Yilmaz: designed the study and wrote the paper. Murat Kavas: performed the study. Ays¸e Ersev: analyzed the data and contributed important reagents. Gulbanu Horzum Ekinci: collected the data. Osman Haciomeroglu and Cansel Atinkaya: contributed important reagents. Esen Akkaya: designed the study and analyzed the data. Conflict of interest The authors have stated explicitly that there are no conflicts of interest in connection with this article.

Introduction Hemangiomas are benign, richly vascular tumors (1). They are proliferative lesions characterized by increased endothelial cell turnover, usually appearing after birth, growing rapidly and then involuting over time (2). Usually hemangiomas are found in subcutaneous tissue, liver, bone, central nervous system and spleen (2, 3). Their occurrence in the mediastinum is extremely rare, with an incidence of less than 0.5% of all mediastinal tumors (4). Hemangiomas are classified as capillary, cavernous or venous types, according to the size of their vascular spaces. Venous hemangiomas

The Clinical Respiratory Journal (2014) • ISSN 1752-6981 © 2014 John Wiley & Sons Ltd

are extremely rare and they account for 1% of all mediastinal hemangiomas (1). We report a case of venous hemangioma in the anterior mediastinum, and a review of the literature concerning the clinical and pathological features of this disease.

Case presentation A 61-year-old Turkish man presented with a 2month history of dyspnea. He was nonsmoker and his medical history was unremarkable. Posteroanterior chest X-ray showed no abnormalities (Fig. 1). His

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Venous hemangioma of the mediastinum

Figure 1. Chest X-ray shows no abnormalities.

physical examination and laboratory values were normal. Computed tomography scan of the thorax showed a 2.5 × 3.2 × 2 cm mass in the anterior mediastinum (Fig. 2). Magnetic resonance imaging revealed a heterogeneous mass of predominantly indeterminate signal intensity. As precise diagnosis is essential to guide further management, the patient underwent a median sternotomy for biopsy and removal of the

Yilmaz et al.

Figure 3. Microscopic examination shows thick-walled, large vascular spaces filled with erythrocytes surrounded by fibrous stroma (hematoxylin and eosin, ×4).

mass. Intraoperative examination showed a brownish mass with a fibrous capsule in the anterior mediastinum. The tumor was easily dissected free from the surrounding structures, and was subsequently resected. Postoperative course was uneventful, and he was discharged to home 4 days after the operation. Macroscopically, the tumor surface was irregular with a brownish-red color giving the impression of a vascular lesion. Its size was 2.8 × 2.5 × 2 cm. Microscopically, hematoxylin and eosin staining showed various size and shape, and thick-walled vascular structures filled with erythrocytes that were located in loose fibrous stroma. No signs of malignancy were noted (Fig. 3). Immunohistochemical staining of the vascular lining cells was positive for CD-34 (Fig. 4). Based on these findings, the tumor was diagnosed as venous hemangioma. There was no evidence of recurrence at the 12 months after surgery.

Discussion

Figure 2. Computed tomography scan of the thorax shows a 2.5 × 3.2 cm mass in the anterior mediastinum.

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Mediastinal hemangiomas are rare tumors, and previous reports indicate that they account for 0.5% or less of all mediastinal tumors (1–6). In 1914, Shennan reported the first case of mediastinal hemangioma (1, 4). According to Chan et al., only 125 welldocumented cases of mediastinal hemangioma had been reported in the literature up until 2009 (7). Hemangiomas are categorized according to the size of their vascular spaces as capillary, cavernous or venous types (1, 7). Over 90% of cases are hemangiomas of either the cavernous or capillary type. Venous

The Clinical Respiratory Journal (2014) • ISSN 1752-6981 © 2014 John Wiley & Sons Ltd

Yilmaz et al.

Figure 4. Endothelial cells lining vascular spaces show positive staining for CD-34 (immunohistochemical staining, CD-34, ×10).

Venous hemangioma of the mediastinum

only 2 of 61 cases (10). Differential diagnosis of venous hemangiomas include other mediastinal masses (1, 6). Surgery is performed for the purpose of diagnosis and treatment (1). Video-assisted thoracic surgery and sternotomy have been performed to extract the tumor (1–4). Because the tumor may recur after incomplete excision, complete excision of the tumor is recommended (5). Our patient underwent sternotomy for diagnosis and treatment. He had no evidence of recurrence 12 months after operation. In conclusion, hemangioma is a very rare lesion of mediastinum. Preoperative diagnosis of mediastinal hemangiomas is difficult with imaging studies. The possibility of hemangioma should be considered in differential diagnosis of mediastinal masses. The treatment of choice for mediastinal hemangioma is surgical complete resection.

References hemangiomas are extremely rare and they account for 1% of all mediastinal hemangiomas (1). Mediastinal hemangiomas can arise anywhere within the mediastinum. In 68% of cases, the anterior mediastinum is involved. The posterior mediastinum is the primary site of involvement in 22% of cases (1). Mediastinal hemangiomas can affect all age groups, but 50% of cases occur in patients younger than 20 years of age (7). There is no sex predilection (1). One half of patients have no symptoms at presentation. The remainder present with nonspecific symptoms, such as cough, chest pain, dyspnea and back pain. Rarely, dysphagia, superior vena cava syndrome and neurologic symptoms can result from a direct pressure effect or invasion of adjacent structures (1, 4, 7). We reported a 61-year-old man patient with mediastinal venous hemangioma. The tumor in the current case was located in the anterior mediastinum. He presented with a 2-month history of dyspnea. There was no symptom because of compression or invasion of adjacent structures in the present case. Because symptoms and radiological findings are usually nonspecific, preoperative precise diagnosis of mediastinal hemangiomas is difficult. Although computed tomography scan and magnetic resonance imaging of the thorax may be useful for diagnosis, characteristic findings of them are poor (1, 8, 9). It was reported that preoperative diagnosis was correct in

The Clinical Respiratory Journal (2014) • ISSN 1752-6981 © 2014 John Wiley & Sons Ltd

1. Yoshino N, Okada D, Ujiie H, et al. Venous hemangioma of the posterior mediastinum. Ann Thorac Cardiovasc Surg. 2012;18: 247–50. 2. Tan C, Alphonso N, Anderson D, Austin C. Mediastinal hemangiomas in children. Eur J Cardiothorac Surg. 2003;23: 1065–7. 3. Schreiner W, Sirbu H. Video-assisted technique for resection of a large mediastinal hemangioma. Multimed Man Cardiothorac Surg. 2010; doi: 10.1510/mmcts. 2009.004135. 4. Abe K, Akata S, Ohkubo Y, et al. Venous hemangioma of the mediastinum. Eur Radiol. 2001;11: 73–5. 5. Mizutani E, Morita R, Kitamura S. Arteriovenous hemangioma in the middle mediastinum: report of a case. Surg Today. 2011;41: 846–8. 6. Hirai K, Takeuchi S, Bessho R, Ohaki Y, Koizumi K, Shimizu K. Venous hemangioma of the anterior mediastinum. J Nippon Med Sch. 2010;77: 115–8. 7. Chan APH, Wong RHL, Wan IYP, Hsin MKY, Underwood MJ, Yim APC. Video-assisted thoracic surgery excision of mediastinal hemangioma. Asian Cardiovasc Thorac Ann. 2009;17: 522–4. 8. Odaka M, Nakada T, Asano H, et al. Thoracoscopic resection of a mediastinal venous hemangioma: report of a case. Surg Today. 2011;41: 1455–7. 9. Agarwal PP, Seely JM, Matzinger FRK. Case 130: mediastinal hemangioma. Radiology. 2008;246: 634–7. 10. Yamazaki A, Miyomoto H, Saito Y, Matsuzawa H, Sakao Y, Anami Y. Cavernous hemangioma of the anterior mediastinum: case report and 50-year review of Japanese cases. Jpn J Thorac Cardiovasc Surg. 1994;193: 221–4.

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Venous hemangioma of the anterior mediastinum.

Mediastinal hemangiomas are rare tumors, with an incidence of 0.5% or less. We herein present a case of venous hemangioma in the anterior mediastinum...
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