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J Thorac Cardiovasc Surg 2006;132:1239-1240
© 2006 The American Association for Thoracic Surgery
Brief Communication |
Department of Thoracic Surgery, Guy's Hospital, London, United Kingdom.
Received for publication April 1, 2006; revisions received April 3, 2006; accepted for publication May 17, 2006. * Address for reprints: Loïc Lang-Lazdunski, MD, PhD, FRCS, Department of Thoracic Surgery, Guy's Hospital, St Thomas St, London SE1 9RT, United Kingdom. (Email: loic.lang-lazdunski{at}gstt.nhs.uk).
Extralobar sequestration (ELS) is a rare congenital lesion of the lung. We report a case of ELS with concurrent absence of left pericardium presenting in an adult as an anterior mediastinal mass.
A 32-year-old man presented with a 3-week history of anterior chest discomfort. He denied any weight loss, night sweats, or fever. Clinical examination was unremarkable. Chest radiography and computed tomography (CT) revealed a cystic mass in the left anterior mediastinum (80 x 80 x 70 mm) in close proximity to the pulmonary trunk (Figure 1). Tumor markers, including 
-fetoprotein, ß-human chorionic gonadotropin, and lactate dehydrogenase, were normal. A bronchogenic cyst or a thymic cyst was suspected. A CT-guided biopsy was performed but was inconclusive. The patient was referred to our department for biopsy or excision of the lesion.
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Retrospective analysis of the chest CT scan by a radiologist revealed a probable feeding vessel at the site of hemorrhage (Figure 1).
Histopathology demonstrated that the lesion had a wall made of bronchial tissue, with mucous glands and cartilage (Figure 2). The cyst was in lung parenchyma and invested by its own pleura. There were also small cysts and lung parenchyma (with airways containing calcified secretions) present within the tissue. There was no evidence of malignancy and no heterologous elements to suggest a dermoid cyst. The diagnosis was of an ELS.
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Pulmonary sequestration is a developmental defect consisting of variable amounts of nonfunctioning lung components that do not communicate with the tracheobronchial tree. It is distinguished from a bronchogenic cyst by the presence of lung parenchyma and also a dedicated blood supply. Pulmonary sequestrations have been classified into intralobar sequestration (ILS), surrounded by normal lung visceral pleura, and ELS, which has its own pleural investment. ILS is at least 3 times more common than ELS.1
ELS is 4 times more common in male patients than in female patients, usually occurs on the left (90%), and usually occurs in the posterior cardiophrenic angle. Cases of ELS have also been reported in the mediastinum and diaphragm or rarely in a subdiaphragmatic position. Unlike ILS, ELS is frequently associated with other congenital anomalies, especially diaphragmatic hernias and foregut malformations. ELS usually presents in infants, although a few adult cases have been reported.2,3 There is only one previous report of ELS with absence of left pericardium in an adult.4
The arterial blood supply of ELS is predominantly from systemic arteries, usually the aorta, but arises from the pulmonary artery in less than 5% of cases.5 The venous drainage is through systemic veins, most often the azygos veins, hemiazygos veins, or inferior vena cava, although there is sometimes partial drainage through the pulmonary veins.4,5
In this case the mass appeared to have blood supply from the LPA. Venous drainage was not identified.
Surgical resection of pulmonary sequestration is the treatment of choice in symptomatic cases and provides tissue for diagnosis. In this case videothoracoscopic excision was not considered because of the large size of the mass and the absence of diagnosis. A 5-cm minithoracotomy was considered a good alternative and allowed easy dissection of the mass. The incision was enlarged into a 10-cm anterior thoracotomy to control bleeding and provide good exposure of the pulmonary hilum.
Because bronchogenic cysts do not contain lung parenchyma in their walls, it was concluded that this mass was an ELS. This hypothesis was reinforced by the finding of a feeding vessel, which is uncommon in foregut cysts. The presence of a feeding vessel obviously confers a higher risk of bleeding during resection of these lesions compared with excision of foregut cysts. Despite careful review of chest CT scans, we did not establish the diagnosis preoperatively. ELS, and more generally pulmonary sequestration, should therefore be considered in the differential diagnosis of anterior mediastinal masses in adults, and the presence of feeding vessels should be actively sought preoperatively and during excision of such lesions.
References
This article has been cited by other articles:
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  M. B. Shadmehr, H. R. Jamaati, B. Saidi, M. Tehrai, and M. Arab Extralobar sequestration in anterior mediastinum with pericardial agenesis. Ann. Thorac. Surg., July 1, 2009; 88(1): 291 - 293. [Abstract] [Full Text] [PDF]
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