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J Thorac Cardiovasc Surg 2005;130:610-612
© 2005 The American Association for Thoracic Surgery
Brief Communication |
a University Hospital for Lung Diseases "Jordanovac,", Clinical Hospital Center "Zagreb," Zagreb, Croatia
b Department of Pathology, Clinical Hospital Center "Zagreb," Zagreb, Croatia
Received for publication November 19, 2004; accepted for publication December 20, 2004. * Address for reprints: Marko Jakopovic, University Hospital for Lung Diseases "Jordanovac," Jordanovac 104, 10000 Zagreb, Croatia (Email: mjakopovic2001{at}yahoo.com).
Bronchogenic cysts are thought to originate from abnormal budding of the ventral foregut. The majority are located in the mediastinum, usually in the subcarinal or paratracheal region. Approximately 15% are reported to arise in the lung. Other locations, such as the pleura, pericardium, or diaphragm, are rare. Bronchogenic cysts are lined by pseudostratified, columnar, ciliated (respiratory) epithelium and may contain serous fluid, mucus, blood, or purulent material. These cysts usually occur in adults but may occur in any age group. Patients usually remain asymptomatic, even though infection or bleeding produces symptoms in up to two thirds of cases. Symptoms associated with these cysts are much more common in children, in whom large cysts may compress the airways with resultant atelectasis, pneumonia, or air trapping.
We report a rare case of carcinoma arising in a bronchogenic cyst.
Clinical Summary
A 40-year-old woman with no medical history was admitted to the hospital because of radiographically proven complete pneumothorax of the right lung. Two weeks before the admission, the patient felt sudden pain in the right thorax spreading to the right shoulder with dyspnea associated with minimal physical activity. The patient’s condition was growing worse with progressive dyspnea, and she felt a lack of air even during rest. She reported to the emergency department where a chest radiograph was performed, showing complete pneumothorax of the right lung. Arterial partial oxygen pressure before admission was 51 mm Hg. Other laboratory findings were in the normal range. On the first day of admission, thoracic drainage was performed. A control chest radiograph was performed on the same day, which showed fast and almost complete reexpansion of the lung parenchyma followed by reexpansion interstitial edema. A cavity with a thin wall approximately 4 cm in diameter was visible on the same chest x-ray film. Lung bulla was suspected, so alpha1-antitrypsin was performed, which showed normal results (1.95 g/L, range 1.2–2.2 g/L). The patient’s condition was improving, so the intercostal tube was removed after 4 days. The control chest radiograph showed complete reexpansion of the lung parenchyma. A cystic lesion of the middle lobe of the right lung was still present. There was massive improvement in arterial oxygen pressure (96 mm Hg), and the patient was discharged from the hospital 5 days after admission. A computed tomographic scan was performed 5 days after discharge. The computed tomographic scan showed a multilocular cystic lesion with a maximal diameter of 4.9 cm and a thin wall located in the center of the middle lobe of the right lung (Figure 1). Surgical resection was indicated, and the patient was readmitted to the hospital 2 weeks after the discharge. Serology for echinococcus was also performed, which showed negative results. Even without the suspicion of malignancy, fiberbronchoscopy was performed, which showed the normal appearance of the bronchi. Tumor markers (CYFRA 21-1 and neuron-specific enolase) were also performed before surgery and were within the limits of those in a healthy subject.
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Pathologic findings showed a multilocular cyst in the middle of the right lobe with a maximal diameter of 4 cm and cavities ranging from 0.3 to 1.5 cm. Histologically, the cyst was lined by ciliated columnar epithelium, and the wall contained areas of fibroelastic tissue, smooth muscle cells, and hyaline cartilage. The cyst wall contained foci of large, atypical epithelial cells—large cell carcinoma (Figure 2). The cyst wall was intact. Bronchopulmonary lymph nodes were not affected.
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Discussion
The first bronchogenic cyst was reported by Meyer
1
in 1859. Since then, there have been numerous reports in the literature describing bronchogenic cysts. Most bronchogenic cysts originate from the mediastinum with a smaller percentage occurring in the pulmonary parenchyma. However, malignant transformation of a bronchogenic cyst is rare. The first report of malignancy associated with a bronchogenic cyst was by Moersch and Clagget
2
in 1947. Since then, only few well-documented cases of malignancy in intrapulmonary bronchogenic cysts have been reported in the English literature.
3–6
To the best of our knowledge, this was the first case of a large cell carcinoma arising in an intrapulmonary bronchogenic cyst. Other reported cases described bronchioalveolar carcinoma, adenocarcinoma, and squamous cell carcinoma.
3–6
There is not enough evidence about carcinogenesis in a bronchogenic cyst. Some evidence suggests that unstable epithelial cells in the cyst wall could have malignant potential and lead to carcinoma. Even though bronchogenic cysts are mainly asymptomatic in adults, St. Georges and colleagues
7
suggested that all visible bronchogenic cysts should be completely resected because the majority of them will eventually become symptomatic or complicated. As shown in our case, some bronchogenic cysts have the potential of malignant transformation.
In conclusion, we believe that complete resection of any bronchogenic cyst is justified because of the minimal risk of malignant transformation and the much greater risk of cyst-related complications (eg, pneumothorax in our case).
References
ber angeborene blasige Missbildung der Lungen, nebst einigen Bemerkungen 
ber Cyanose aus Lungenleiden. Arch Patol Anat. 1859;16:78-95.
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