(Arch Pathol Lab Med. 1998;122:915-919)
Epithelial malignancy is known to arise in association with cystic diseases of the lung. In most reported cases in which the type of cyst was described, the underlying cystic diseases were various congenital cysts or bullous lesions of the lung.1-12 Recently, we examined an autopsy case of lung carcinoma arising in a placentoid bullous lesion of the lung (PBLL)13-15 in a 52-year-old man. Placentoid bullous lesion of the lung is an unusual cystic lesion whose histogenesis has not yet been sufficiently established. This rare tumor was studied histologically and immunohistochemically.
REPORT OF A CASE
A 52-year-old man was admitted on November 30, 1995, for further evaluation of cervical lymphadenopathy and an abnormal shadow on chest roentgenogram. The abnormal shadow had first been noted by a family physician 6 years before admission. Computed tomography at that time indicated that the shadow represented a giant bulla in the right middle lobe. No findings suggestive of malignancy were found. The patient remained well until cervical lymphadenopathy was noted. The patient had a history of treatment with antituberculous drugs for pulmonary tuberculosis at the age of 9. He had smoked 25 cigarettes a day for 35 years.
Results of radiologic, pathologic, and laboratory examinations on and after admission were as follows. Chest roentgenogram demonstrated a large homogeneous shadow with a sharp border in the middle field of the right lung (Figure 1). Computed tomography showed a cystic lesion with an inner low-density area (Figure 2). A percutaneous biopsy specimen of the cystic lesion suggested a diagnosis of adenocarcinoma, and biopsy of a cervical lymph node revealed metastatic adenocarcinoma. A ^sup 99m^Tc bone scintigraphy also suggested metastases to the thoracic vertebrae and left parietal bone. Results of routine blood examination on admission were within normal limits. Only the serum level of CA125 was moderately elevated.
Although neck irradiation was performed for relief of pain and an anticancer drug (doxorubicin) was injected locally into the right pleural space to counter gradual accumulation of pleural fluid, systemic chemotherapy could not be given because of the patient's poor general condition. The patient died of respiratory insufficiency due to massive pleural effusion and pulmonary edema 4 months after admission. Autopsy was performed approximately 10 hours after death.
AUTOPSY FINDINGS
The left lung weighed 400 g, and the right weighed 680 g. The right pleura demonstrated marked thickening and fibrinous adhesion due to pleuritis carcinomatosa. There was also a fluid-filled loculated area in the pleural space. The surface of the pleura of the left lung was almost completely intact macroscopically, but microinvasion of the pleura by carcinoma cells was found histologically. Pleural fluid compressing the lungs measured 1500 mL on the left and 600 mL on the right. The lungs were edematous, but were almost completely expanded.
The central portion of the middle lobe with atelectasis was occupied by a sharply defined cystic tumor, measuring 4.5 x 3.0 x 2.5 cm, which was away from the loculated area mentioned above. The cyst contained a soft villous mass mimicking placental chorionic villi of early gestation, which was partially attached to the slightly shaggy inner surface of the thin wall (Figure 3). Although the bronchi of the middle lobe ran near the tumor, they were not connected to it. Microscopically, the mass was composed of papillary structures with round, oval, and trabecular shapes similar to chorionic villi at low magnification (Figure 4). The mass was a papillary adenocarcinoma with a wide stromal core. Carcinoma cells lining the stroma showed round, oval, or occasionally irregularly shaped and vesicular nuclei with relatively abundant eosinophilic cytoplasm (Figure 5). The apical margins of carcinoma cells were stained with Alcian blue. Most of the cyst wall was also covered with carcinoma cells. It was peculiar that the wide stromal core of the papillary structures comprised fat cells, infiltrating or aggregated lymphocytes, foam cells, and smooth muscle cells as well as vascular channels that frequently showed mural hyalinization (Figure 6). Preexisting cuboidal epithelial cells without atypia were seen on the surface of the papillary structures and in some parts of the cyst wall (Figure 7). The macroscopic and microscopic placenta-like appearance and the characteristic features of the stroma of the papillary structures were very similar to those of PBLL in areas not composed of adenocarcinoma. Therefore, the structure may be interpreted as carcinoma cells spreading superficially in PBLL, resulting in a papillary growth pattern. The carcinoma invaded the cyst wall and the surrounding lung tissue. Invasion of blood and lymphatic vessels was seen frequently.
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