Ann Thorac Surg 2014;97:687–9

References 1. Risher WH, Arensman RM, Ochsner JL. Congenital bronchoesophageal fistula. Ann Thorac Surg 1990;49:500–5. 2. Zhang BS, Zhou NK, Yu CH. Congenital bronchoesophageal fistula in adults. World J Gastroenterol 2011;17:1358–61. 3. Braimbridge MV, Keith HI. Oesophago-bronchial fistula in the adult. Thorax 1965;20:226–33. 4. Kim JH, Park KH, Sung SW, Rho JR. Congenital bronchoesophageal fistulas in adult patients. Ann Thorac Surg 1995;60: 151–5. 5. Smith DC. A congenital broncho-oesophageal fistula presenting in adult life without pulmonary infection. Br J Surg 1970;57:398–400. 6. Marco C, Doncel F, Veloso E, Viver JM, Vidal J. Non-surgical closure of a benign oesophagobronchial fistula. Br J Surg 1986;74:415. 7. Shimada T, Abo S, Kitamura M, Hashimoto M, Shikama T, Kimura Y. A case of congenital esophagobronchial fistula communicated between esophageal diverticulum and left main bronchus in the adult–a review of 47 cases in the Japanese literature [Article in Japanese]. Nihon Kyobu Geka Gakkai Zasshi 1992;40:2102–6.

Cardiac Cavernous Hemangioma and Multiple Pulmonary Cavernous Hemangiomas Lili Yang, PhD, Jun Dai, PhD, Ying Xiao, M. Med, Henghui Cheng, PhD, and Qiurong Ruan, PhD Institute of Pathology of Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, China

We describe for the first time a rare coexistence of a cardiac cavernous hemangioma with multiple pulmonary cavernous hemangiomas. Computed tomography revealed bilateral pulmonary nodules, left pleural effusion, and pericardial effusion. Positron emission tomography showed a pericardial neoplasm. Pathologically, multiple large dilated vascular spaces, lined by a single layer of endothelial cells and filled with blood, were revealed in both the cardiac tumor and the pulmonary nodules. Immunohistochemical examination of the lining cells showed positivity for CD31, FLI1, FVIII, and CD34. Taken together, these findings led to the diagnosis of cardiac cavernous hemangioma and multiple pulmonary cavernous hemangiomas. (Ann Thorac Surg 2014;97:687–9) Ó 2014 by The Society of Thoracic Surgeons

Accepted for publication May 15, 2013. Address correspondence to Dr Ruan, Institute of Pathology, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan 430030, China; e-mail: [email protected].

Ó 2014 by The Society of Thoracic Surgeons Published by Elsevier Inc

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avernous hemangiomas are characterized by sharply defined yet unencapsulated masses, which are composed of large dilated vascular spaces lined by a single layer of endothelial cells and filled with blood. They are benign vascular tumors that are frequently located in the skin, subcutaneous tissues, and liver. Cavernous hemangiomas can occur in various internal organs, but they rarely occur in the heart and lungs. We report a rare case of a co-occurring cardiac cavernous hemangioma and multiple pulmonary cavernous hemangiomas (PCHs) and review the literature. A 36-year-old man with a 1-month history of pericardial effusion was admitted to our hospital. Imaging with ultrasonography and chest roentgenography (Fig 1A) showed bilateral pleural effusion and pericardial effusion. Computed tomography (CT) of the chest and heart (Figs 1B, 1C) showed bilateral pulmonary nodules, left pleural effusion, and pericardial effusion. Positron emission tomography showed a pericardial neoplasm. After extensive workup, a thoracotomy, pericardial tumor biopsy, and right lung biopsy were performed. A dark red, easily bleeding cystic tumor, adherent to the right atrium and measuring 8  6 cm, was identified in the pericardial cavity. It could not be separated as a result of myocardial invasion. A small edge was removed for pathologic examination. The right mediastinal pleura was opened, and several nodules were found in the subpleural lung tissue. Some of this tissue was also removed for pathologic examination. The samples were fixed in 10% buffered formalin, embedded in paraffin, sectioned conventionally, and then stained with hematoxylin and eosin. CD31, FLI1, FVIII, CD34, calretinin, WT-1, D2-40, smooth muscle actin, desmin, and caldesmon were detected using immunohistochemical staining methods. Gross examination revealed the cardiac neoplasm to be a dark red lamellar mass measuring 2  2  1 cm; it was soft and bled easily. The pulmonary tissue was a dark brown mass measuring 2.5  2  1 cm and contained multiple sharply defined but not encapsulated nodules. The cut surface showed focal hemorrhagic and cystic areas. Microscopically, multiple irregular dilated vascular spaces lined by a single layer of endothelial cells were observed in the cardiac tumor (Fig 2A). Similarly, several nodules composed of large dilated vascular spaces were scattered in the lung tissue; these were variably filled with blood. The spaces were lined by a single layer of thin endothelial cells (Figs 2B, 2C). Immunohistochemical examination of the lining cells revealed positivity for CD31 (Fig 2D), FLI1, FVIII, and CD34, consistent with a lesion of endothelial origin. In addition, some dilated thick vascular walls in the pulmonary nodules were positive for smooth muscle actin. The results of staining for calretinin, WT-1, D2-40, desmin, and caldesmon were negative.

Comment Hemangiomas are benign tumors that are mainly composed of blood vessels. The histologic classification 0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2013.05.111

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Dr Lerut was a visiting professor at the Department of Thoracic Surgery, Marmara University Faculty of Medicine supported by TUBITAK (The Scientific and Technological Research Council of Turkey) program 2221.

CASE REPORT YANG ET AL CARDIAC CAVERNOUS HEMANGIOMA AND PCHS

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CASE REPORT YANG ET AL CARDIAC CAVERNOUS HEMANGIOMA AND PCHS

Ann Thorac Surg 2014;97:687–9

Fig 1. (A) Chest roentgenogram showing bilateral pleural effusion and pericardial effusion. (B, C) Computed tomographic scan of the chest and heart showing bilateral pulmonary nodules, left pleural effusion, and pericardial effusion.

FEATURE ARTICLES

includes capillary, cavernous, and arteriovenous hemangiomas, but the cause is not yet very clear. Cardiac hemangiomas are exceedingly rare, representing only 1% to 2% of all detected benign heart neoplasms [1]. They can occur at any age and in any layer of the heart, including the epicardium, myocardium, and endocardium [2], although the epicardium is the most common location [3]. Cardiac hemangiomas can also develop in all the cardiac chambers, but they mainly occur on the right side of the heart and the left atrium [2]. Pulmonary cavernous hemangiomas (PCHs) are also exceptionally rare. Fig 2. (A) Multiple irregular dilated vascular spaces lined by a single layer of endothelial cells in the cardiac tumor. (Hematoxylin and eosin; 40.) (B, C) Several nodules composed of multiple irregular dilated vascular spaces scattered in the lung tissue. (Hematoxylin and eosin; 40.) (D) The lining cells of the dilated vascular spaces showed CD31 positivity (Immunohistochemical examination; 40.)

Generally, PCHs are solitary lesions and can present in all lobes, involving both lung parenchyma and subpleural tissue [4]. The diagnosis in our patient was cardiac cavernous hemangioma and multiple PCHs. To our knowledge, no such case has ever been reported previously. Most hemangiomas are found incidentally. The symptoms of cardiac hemangiomas depend on the location and size of the tumors. Patients may have no symptoms, or they may present with dyspnea, heart palpitations, atypical chest pain, or arrhythmia or pericardial effusion. [2]. Similarly, some patients with PCHs have no obvious

symptoms, whereas others display a series of clinical processes such as hemoptysis and respiratory distress until heart failure occurs [4]. Echocardiography is usually the initial imaging examination for the diagnosis of cardiac tumors, although computed tomography and magnetic resonance imaging actually have a higher accuracy rate in exposing this condition. Usually, these modalities have been used in preoperative diagnosis [5]. However, the nature of the tumors cannot be cleared by preoperative diagnosis in many cases. Preoperative diagnosis of PCHs is also difficult. All cases that have been reported were diagnosed by intraoperative or postoperative examination or at autopsy. The histologic appearances of cardiac cavernous hemangiomas and PCHs are no different from the appearances of other parts. The diagnosis in our patient was not difficult based on the histologic appearance and immunohistochemical reactivity. However, other entities, such as mesothelioma, angiosarcoma, and myxoma, must sometimes be considered in the differential diagnosis. Microscopically, most epithelioid mesotheliomas show bland cells and little mitosis. Epithelioid tumor cell nests can be seen within a bland fibrous stroma [6]. Calretinin, WT-1, and D2-40 should be positive by immunohistochemical examination. Mitotic activity, cellular pleomorphism, necrosis, and cellularity can distinguish hemangiomas from angiosarcomas. Vascular endothelial markers such as FVIII, CD31, and CD34 all have a strong reaction in angiosarcomas [7]. Myxomas have a typical appearance, with spindle cells widely spaced by abundant myxoid matrix, which can differentiate them from cavernous hemangiomas. Hemangiomas may have a good prognosis and a low recurrence rate. So far, no malignant change has been reported to our knowledge. Total resection should be the standard treatment for symptomatic cavernous hemangiomas whenever possible [8]. For most cardiac cavernous hemangiomas, complete excision is possible. However, some lesions that have extensive infiltration are appropriate only for incomplete resection or simple biopsy, and the prognosis is usually poor [2]. Similarly, solitary PCHs should be treated with surgical resection, usually wedge resection or enucleation [4]. However, no intervention was undertaken in the present reported case of multiple PCHs. In this case, cavernous hemangiomas were found in both the heart and the lungs, which was exceedingly rare and complicated. Embolizing the remaining cavernous hemangiomas was considered; however, the patient’s condition did not allow it. Close radiographic and clinical follow-up was needed. Unfortunately, as a result of pericardial effusion and pleural effusion, the patient experienced a series of processes such as heart failure, respiratory failure, causally insufficient blood supply, and ischemia and anoxia of the vital organs, and finally died of multiple organ dysfunction syndrome 2 months later.

CASE REPORT LIU ET AL RUPTURE OF CYSTIC TERATOMA WITH SVC SYNDROME

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2. Tabry IF, Nassar VH, Rizk G, Touma A, Dagher IK. Cavernous hemangioma of the heart: case report and review of the literature. J Thorac Cardiovasc Surg 1975;69:415–20. 3. Botha J, Ihlberg L, Elhenawy A, et al. A giant cavernous hemangioma of the heart. Ann Thorac Surg 2010;90:293–5. 4. Fine SW, Whitney KD. Multiple cavernous hemangiomas of the lung: a case report and review of the literature. Arch Pathol Lab Med 2004;128:1439–41. 5. Oshima H, Hara M, Kono T, Shibamoto Y, Mishima A, Akita S. Cardiac hemangioma of the left atrial appendage: CT and MR findings. J Thorac Imaging 2003;18:204–6. 6. Butnor KJ, Nicholson AG, Allred DC, et al. Expression of renal cell carcinoma-associated markers erythropoietin, CD10, and renal cell carcinoma marker in diffuse malignant mesothelioma and metastatic renal cell carcinoma. Arch Pathol Lab Med 2006;130:823–7. 7. Haber MH, Gattuso P, Spitz D, David O. Differential diagnosis in surgical pathology. Philadelphia: Saunders Company; 2002:24. 8. Chen X, Lodge AJ, Dibernardo LR, Milano CA. Surgical treatment of a cavernous haemangioma of the heart. Eur J Cardiothorac Surg 2012;41:1182–3.

Spontaneous Rupture of a Cystic Mediastinal Teratoma Complicated by Superior Vena Cava Syndrome Chia-Hsin Liu, MD, Yi-Jen Peng, MD, PhD, Hong-Hau Wang, MD, Yeung-Lung Cheng, MD, PhD, and Chien-Wen Chen, MD Division of Pulmonary and Critical Care, Department of Internal Medicine, and Departments of Pathology and Radiology, and Division of Thoracic Surgery, Department of Surgery, Tri-Service General Hospital, National Defense Medical Center, Taipei, Taiwan

Spontaneous rupture of cystic mediastinal teratomas is rare but may cause serious complications. Here we report an unusual case of a cystic teratoma, which ruptured into the mediastinal and pleural cavities resulting in superior vena cava syndrome, acute mediastinitis, and pleural effusion. Early diagnosis and prompt surgical treatment of ruptured mediastinal teratomas are essential to preventing life-threatening complications. (Ann Thorac Surg 2014;97:689–91) Ó 2014 by The Society of Thoracic Surgeons

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eratomas, which contain ectodermal, mesodermal, and endodermal tissue remnants [1] are the most common tumor type in the anterior mediastinum. Usually, teratomas are asymptomatic and are discovered incidentally by chest radiography or computed tomography (CT). On rare occasions, a cystic teratoma

Accepted for publication June 3, 2013.

References 1. Kipfer B, Englberger L, Stauffer E, Carrel T. Rare presentation of cardiac hemangiomas. Ann Thorac Surg 2000;70:977–9. Ó 2014 by The Society of Thoracic Surgeons Published by Elsevier Inc

Address correspondence to Dr Chen, Division of Pulmonary and Critical Care, Department of Internal Medicine, Tri-Service General Hospital, National Defense Medical Center, 325, Cheng-Gung Rd, 2nd Section, Taipei, 114, Taiwan; e-mail: [email protected].

0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2013.06.112

FEATURE ARTICLES

Ann Thorac Surg 2014;97:689–91

Cardiac cavernous hemangioma and multiple pulmonary cavernous hemangiomas.

We describe for the first time a rare coexistence of a cardiac cavernous hemangioma with multiple pulmonary cavernous hemangiomas. Computed tomography...
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