Ann Thorac Surg 2000;69:933-935
© 2000 The Society of Thoracic Surgeons
Case Reports
Bronchioloalveolar carcinoma arising in a bronchogenic cyst
Chiaki Endo, MDa,b,
Tadashi Imai, MDa,
Hideyuki Nakagawa, MDa,
Akio Ebina, MDa,
Mitsuomi Kaimori, MDb
a Department of Respiratory Diseases, Aomori Prefectural Central Hospital, Aomori, Japan
b Department of Pathology, Aomori Prefectural Central Hospital, Aomori, Japan
Address reprint requests to Dr Endo, 16-5-102 Ikenodai, Kohriyama 963-8875, Japan
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Abstract
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We report the case of a 37-year-old woman with a radiographically cystic lung lesion. Lobectomy was performed. Histopathologic examination showed a bronchioloalveolar carcinoma arising in a bronchogenic cyst. This suggests that epithelial cells of bronchogenic cysts can undergo malignant transformation. It may be prudent to recommend complete resection of any bronchogenic cyst.
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Introduction
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Bronchioloalveolar carcinoma is a subtype of pulmonary adenocarcinoma occurring as an isolated nodule, multiple nodules, or a relatively diffuse lesion. Cavitation in bronchioloalveolar carcinoma is unusual. We report a rare case of a bronchioloalveolar carcinoma arising in a bronchogenic cyst.
A 37-year-old woman with no history of smoking had no previous medical history before a radiologic lung abnormality was found in an industrial medical examination. Computed tomographic scans showed a cystic lesion in the right lung (Fig 1). Right posterolateral thoracotomy on July 2, 1997 showed a soft cystic lesion at the right lower lobe. A right lower lobectomy was performed with hilar and mediastinal lymphatic dissection.
In the pathology report, the cystic lesion was 2.7 x 2.5 x 2.0 cm with a 1.8-cm cavity. Microscopic examination showed only one cyst; carcinomatous foci composed of large, well-differentiated, mucus-secreting columnar cells lined the intact alveolar septa and the cyst wall. Many alveoli were filled with macrophagic cells and mucus, typical of mucinous bronchioloalveolar carcinoma (Fig 2). Part of the cavity was lined by ciliated respiratory epithelium close to carcinomatous columnar cells (Fig 3). The wall also contained a small cartilage plate and chronic inflammatory infiltration (Fig 4). The conclusion was bronchioloalveolar carcinoma arising in a bronchogenic cyst. One year later, the patient was in good condition, with no signs of metastasis.
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Fig 2. Bronchioloalveolar carcinoma. Well-differentiated mucinous malignant cells grow along alveolar walls (hematoxylin-eosin, x40).
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Fig 3. Ciliated respiratory epithelium close to carcinomatous columnar cells (hematoxylin-eosin, x200).
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Fig 4. Small cartilage plate, fibromuscular connective tissue, and chronic inflammatory infiltration showing the cyst to be a bronchogenic cyst (hematoxylin-eosin, x100).
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Comment
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Cavitation in bronchioloalveolar carcinoma is unusual. Preexisting lung cysts may antedate the development of malignancy, or pseudocavitation may occur due to distended alveoli filled with abundant mucus [1].
Bronchogenic cysts are congenital and can be either extrapulmonary or intrapulmonary [2]. They typically are spherical and unilocular. They are lined by the ciliated columnar epithelium and their walls may also contain cartilage plates, seromucinous glands, and fibromuscular connective tissue. The most reliable criterion of intrapulmonary bronchogenic cysts is thought to be the presence of cartilage in the wall [3].
In our case, the cyst was unilocular, and the wall contained a small cartilage plate. Part of the wall was lined by ciliated columnar epithelium. These findings indicated the cyst was a bronchogenic cyst. Carcinomatous columnar cells were adjacent to ciliated respiratory epithelium lining the wall, suggesting bronchogenic cyst epithelial cells developed into bronchioloalveolar carcinoma.
Stocker described many of bronchogenic cysts representing examples of type 1, congenital cystic adenomatoid malformations (CCAM) [4]. Sheffield and associates, described premalignant changes in type 1 CCAM containing mucus cells [5]. Several case reports showed an association between CCAM and bronchioloalveolar carcinoma [5, 6]. However, in our case, the cyst was unilocular, in contrast to CCAM type 1, which consists of multiple large epithelial-lined cysts. Our cyst was diagnosed as a bronchogenic cyst, a rare case of association between a bronchogenic cyst and bronchioloalveolar carcinoma.
St-Georges and associates recommended all presumed bronchogenic cysts seen in the adult be resected because the majority will ultimately become symptomatic or complicated [2]. Based on our experience, bronchogenic cysts have the potential for malignant changes. In conclusion, it may be prudent to recommend complete resection of any bronchogenic cyst.
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References
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Weisbrod G.L., Towers M.J., Chamberlain D.W., Herman S.J., Matzinger F.R.K. Thin-walled cystic lesions in bronchioloalveolar carcinoma Radiology 1992;185:401-405.[Abstract/Free Full Text]
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St-Georges R., Deslauriers J., Duranceau A., et al. Clinical spectrum of bronchogenic cysts of the mediastinum and lung in the adult. Ann Thorac Surg 1991;52:6-13.[Abstract]
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Gould S.J., Hasleton P.S. Congenital abnormalities. In: Hasleton P.S., ed. Spencer’s pathology of the lung. New York: McGraw-Hill, 1996:69-78.
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Stocker J.T. The respiratory tract. In: Stocker J.T., Dehener L.P., eds. Pediatric pathology. Philadelphia: JB Lippincott, 1992:505-573.
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Sheffield E.A., Addis B.J., Corrin B., McCabee M.M. Epithelial hyperplasia and malignant change in congenital lung cysts. J Clin Pathol 1987;40:612-614.[Abstract/Free Full Text]
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Benjamin D.R., Cahill J.L. Bronchioloalveolar carcinoma of the lung and congenital cystic adenomatoid malformation. Am J Clin Pathol 1991;95:889-892.[Medline]
Accepted for publication July 9, 1999.
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Abstract
Introduction
