Ann Thorac Surg 2006;81:e13-e15
© 2006 The Society of Thoracic Surgeons
Case report
Left Ventricular Bronchogenic Cyst
Xiang Wei, MD
*
,
Alfred Omo, MD,
Tiecheng Pan, MD,
Jun Li, MD,
Ligang Liu, MD,
Min Hu, MD
Department of Cardiothoracic Surgery, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, China
Accepted for publication December 8, 2005.
* Address correspondence to Dr Wei, Department of Cardiothoracic Surgery, Tongji Hospital, Huazhong University of Science and Technology, Wuhan, 430030 China (Email: xiangwee{at}yahoo.com.cn).
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Abstract
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Bronchogenic cysts occurring in the left ventricle are a medical rarity. One successfully operated case is reported herein. The location of the cyst was just between the epicardium and myocardium of the inferior left ventricular wall, adjacent to the apex of the heart. Complete excision was achieved through a left anterolateral thoracotomy without extracorporeal circulation.
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Introduction
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Bronchogenic cysts arise from an abnormal budding of the ventral diverticulum of the foregut or the tracheobronchial tree during embryogenesis and are most commonly primary cysts of the mediastinum. Only a few cases occurring in the heart have been described, but their location in the left ventricle is extremely rare [1. So far we have not found any case in the world literature. Herein we report a case of bronchogenic cyst occurring in the left ventricle of a 2-year-old boy presenting with intermittent precordial pain. Diagnostic methods, surgical management, and histopathologic findings provide a complete overview of this unusual case.
A 2-year-old boy presented to our hospital with a chief complaint of intermittent precordial pain for 1 month. Physical examination was unremarkable and transthoracic echocardiography revealed a cyst that measured 2.0 x 1.5 cm in size, located just beneath the left ventricle, adjacent to the apex of the heart (Fig 1). Magnetic resonance imaging revealed an irregular cystic lesion that measured 2.5 x 1.5 cm in size, located beneath the inferior wall of the left ventricle (Fig 2).
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Fig 1. Transthoracic echocardiography showing an irregular cyst (arrow) located beneath the left ventricle, adjacent to the apex of the heart. (LV = left ventricle.)
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Fig 2. Magnetic resonance imaging showing an irregular cyst (arrow) beneath the inferior wall of the left ventricle. (LV = left ventricle.)
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An impression of intrapericardial cyst was made, and intrapericardial cystectomy was planned subsequently. Through a left anterolateral thoracotomy through the fourth intercostal space, the pericardium was opened above the left phrenic nerve. A multi-cystic tumor arising from the inferior wall of the left ventricle could be seen (Fig 3).
We performed tumor enucleation and managed to resect it from the myocardium. However, because the pedicle of the tumor was rooted inside the myocardium, traction to expose the tumor pedicle led to a small perforation of the left ventricular wall, just before the end of the enucleation. It caused some bleeding from the left ventricle for which 200 mL of pure red blood cells was transfused for compensation. We repaired the perforation using double pledgetted 4-0 Prolene sutures (Ethicon, Somerville, NJ). The patient's postoperative course was uneventful.
Grossly, the excised tumor measured 3.5 x 2.0 x 2.0 cm in size and had multi-cystic features with turbid contents. Microscopically the tumor consisted of thin-walled multiple cysts attached to the resected myocardium. The inner cavities were lined with pseudo-stratified ciliated columnar epithelium and cartilages (Fig 4), which are characteristic features similar to that of normal bronchi and confirm a pathologic diagnosis of bronchogenic cyst.
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Fig 4. Hematoxylin & eosin stained photomicrograph (magnification, x20) showing the cyst was adjacent to the myocardial cells (MC) and lined by respiratory epithelium (RE) and cartilages (C).
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Comment
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Bronchogenic cysts are lesions of congenital origin that arise from an abnormal budding of the ventral diverticulum of the foregut or tracheobronchial tree during embryogenesis. They may occur in the mediastinum and intrapulmonary region, and unusual sites include the neck [2, intrapericardial [3–4, and intraabdominal area [5.
Bronchogenic cysts of the heart are exceedingly rare and usually occur on the epicardial surface or the myocardium with or without projection into one of the cardiac chambers. In our review of world literatures, less than 20 cases were found, of which the majority reported were located in the right side of the heart, including the interatrial septum [3, the right ventricular septum [4, and the right ventricle [6. However none from the left ventricle has been reported. In our case, the bronchogenic cyst located at the inferior wall of the left ventricle was confirmed intraoperatively, and the pedicle of the cyst was rooted inside the myocardium, conforming with the previously mentioned microscopic finding of myocardial cells adjacent to the respiratory epithelium and cartilages. As mentioned by Kawase and colleagues [3, during early fetal development, cardiac primordial exist in a place very near to the foregut or primitive tracheobronchial tree. It is at this period that a bronchogenic cyst of the heart could arise from an abnormal budding of the tracheobronchial tree and aberrantly migrate into a myocardial location.
Bronchogenic cysts are most often symptomatic in childhood; however they are rarely observed in adults. These cysts do present with chest pain in the retrosternal area and with dysphagia, cough, fever, hemoptysis, and recurrent infection. We believe our patient's precordial pain was due to the occupying lesion pressing on the surrounding tissue, rather than from an ischemic origin. Clinical manifestations of cardiac tumors are generally nonspecific and often reflect the chamber of origin rather than the specific tumor type. Transthoracic echocardiography should be the investigation of first choice for patients with suspected cardiac tumor, and it can provide relatively accurate information of tumor size, location, point of attachment, mobility, and hemodynamic relationship, and therefore it can be recommended as an optimal choice for initial investigation and postoperative follow-up. In our case, transthoracic echocardiography reported an irregular shaped, multi-cystic lesion, which was the clue to the diagnosis of the bronchogenic cyst. Magnetic resonance imaging provides not only the same information as echocardiography, but also the spatial relationship between the tumor and the cardiac chambers, which is crucial in planning for surgical intervention [6. However in our case the true location of the cyst was missed; its size reported by the radiologist did not conform with the actual size we measured after excision, and this variation could have been due to slicing of magnetic resonance imaging section in which some portion of the tumor was missed. The limitation of this method also lies in its scarce availability and high costs.
The rationale for resection of all cardiac tumors is not unequivocally clear. The decision for operation is easy to make for symptomatic tumors, as in our case, and asymptomatic left-sided tumors of the heart if there is obstruction to blood flow or interference with valve function. Resection of apparently benign cardiac tumors such as bronchogenic cysts is recommended to avoid potential complications such as cardiac failure, dysrhythmias, conduction disturbances, syncope, sudden cardiac death, or embolization that can be prevented by early resection. Others are to rule out malignancy or diagnose a malignant tumor while it is still resectable. The majority of reported cases were resected completely under cardiopulmonary bypass through a full sternotomy [3, 4, 6. Because in our case the tumor was a multi-cystic lesion without an impression of invasion to any cardiac chamber, we thought a noncardiopulmonary bypass procedure through a left anterolateral thoracotomy could enable an easy tumor resection and avoid the risk of complications associated with cardiopulmonary bypass and sternotomy. However, when the cyst was enucleated completely, traction on its pedicle led to a small perforation of the left ventricular wall, which caused some bleeding that necessitated blood transfusion. Because a bronchogenic cyst of the left ventricle may lie between the epicardium and myocardium as in our case, and could be rooted inside the myocardium as undetectable by magnetic resonance imaging and echocardiography, we suggest that tumor resection through a sternotomy would be much safer because it also provides better access for instituting cardiopulmonary bypass when necessary.
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References
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- Deenadayalu RP, Tuuri D, Dewall RA. Intrapericardial teratoma and bronchogenic cystreview of literature and report of successsful surgery in infant with intrapericardial teratoma. J Thorac Cardiovasc Surg 1974;67:945-952.[Medline]
- Sanli A, Onen A, Ceylan E, Yilmaz E, Silistreli E, Acikel U. A case of a bronchogenic cyst in a rare location Ann Thorac Surg 2004;77:1093-1094.[Abstract/Free Full Text]
- Kawase Y, Takahashi M, Takemura H, Tomita S, Watanabe G. Surgical treatment of bronchogenic cyst in the interartrial septum Ann Thorac Surg 2002;74:1695-1697.[Abstract/Free Full Text]
- Weinrich M, Lausberg HF, Pahl S, Pahl S, Schafer HJ. A bronchogenic cyst of the right ventricular endocardium Ann Thorac Surg 2005;79:e13-e14.[Abstract/Free Full Text]
- Coselli MP, de Ipolyi P, Bloss RS, Diaz RF, Fitzgerald JB. Bronchogenic cyst above and below the diaphragmreport of eight cases. Ann Thorac Surg 1987;44:491-494.[Abstract]
- Prates PR, Lovato L, Homsi-Neto A, et al. Right ventricular bronchogenic cyst Tex Heart Inst J 2003;30:71-73.[Medline]
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Abstract
Introduction
