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Carcinoma Arising in a Congenital Lung Cyst
Carcinoma Arising in a Congenital Lung Cyst*·** Report of a Case LT . STANLEY BAUER, (MC). USNRt Corpus Christi, Texas
In 1941, Womack and Graham" first called attention to the occurrence of squamous metaplasia in the lining of congenital lung cysts. This observation was based upon the study of three cases, which they felt suggested a pathogenetic role for this change in the formation of squamous cell carcinoma. Both prior to and since this article by Womack and Graham, several papersr":":"... have appeared which either cite or report cases of carcinoma associated with lung cysts. However, most of these 1·...... have been cases of independent bronchial carcinoma in a cystic lung; or they have been so poorly documented that no direct relationship was apparent between a carcinoma and a pulmonary cyst. If one accepts the criticism offered by some authors," there are only three cases':":" in the English literature in which there is irrefutable documentation of a carcinoma arising in the wall of a congenital pulmonary cyst. The following case is presented as another example of this occurrence.
Case Report V.RL.: This 30 year-old white man cigarette smoker was known to have a solitary right lower lobe cyst, which had. been detected on a routine physical examination one year prior to admission (Fig. 1). He had received no treatment and had. been asvmp-
FIGURE 1: Chest roentgenogram taken one year prior to admission showing solitary pulmonary cyst in right lower lobe . -From the Department of Pathology, U. S . Naval Hospital. - -The views expressed are those of the author anJ do not necessarily represent the views of the Bureau of Medicine and Surgery, Navy Department. tPresently at the Beth Israel HosPital, New York City. New York. 552
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tomatic until one week before his admission to the U. S. Naval Hospital, when he developed an aching pain in the right posterior aspect of the chest. The physical findings were eSBent1ally normal except for tenderness over the posterior portion of the right sixth through eighth ribs. Chest roentgenograms revealed a single cyst in the superior portion of the right lower lobe. Comparison with the previous film (Fig. 1) showed no change in the size or configuration of the Cyst. Right thoracotomy was performed, at which time the lower lobe was found to be adherent to the seventh rib posteriorly. A frozen section of the lung revealed an oat cell carcinoma. Right lower lobectomy was performed. The surgical specimen disclosed a thin walled spherical cyst located between the posterior basal and lateral basal segments. The cyst measured 8.0 em. In diameter. On the postero-lateral wall of the cyst there was a firm greyish-white tumor nodule which measured 1.0 em. in greatest diameter (Fig. 2) . The tumor 1n1Utrated the adjacent lung, where it had been separated from the rib cage . There were four small communications between the cyst and the bronchial tree ; however, there was no bronchus related to the tumor. All of the major bronchi of the lobe were unremarkable. Microscopic sections revealed the cyst to be lined with typical bronchial epithelium (Fig. 3). The cyst wall was composed of a thin layer of chronically infiamed fibrous tissue and rare smooth muscle elements. The tumor. which arose from the lining of the cyst, was composed of well differentiated squamous cell carcinoma (Fig. 4) mixed with areas of oat cell carcinoma. The tumor inft1trated the surrounding lung tissue and pleura. A hllar lymph node contained metastatic squamous cell carcinoma. The immediate postoperative course was uncomplicated. Because of the local rib invasion and hUar node spread, x-ray and nitrogen mustard therapy were instituted. His condition deteriorated progressively and he expired seven months after admission. Postmortem examination revealed a large tumor mass in the right hemithorax. The tumor 1n1Utrated the third through the eighth vertebral bodies and the adjoining ribs and soft tissues. There were distant metastases in the hilar lymph nodes, both adrenal glands and liver. On microscopic study all of the tumor tissue was found to be composed of poorly differentiated squamous cell carcinoma.
Comment This case bears out the contention of Womack and Graham lS and Graham" that the bronchial epithelium of congenital lung cysts can give rise to squamous cell carcinoma. Perhaps the development of carcinoma should be considered as a PQ881ble complication when planning the management of a case of congenital lung cyst. This consideration might serve as another indication for surgical intervention in these cases.·
FIGURE 2: Lung cyst showing squamous cell carcinoma (arrow) arising in the wall.
554
LT. STANLEY BAUER
Nov. . 1961
Figure 3
Figure 4
FIGURE 3: Section through cyst wall showing typical bronchial epithelium. (H . and E. DOO) . FIGURE 4: Section through tumor mass showing welld1fferentiated squamous cell carcinoma. (H . and E. DOO) . REFERENCES
1 Bass, H. E ., and Singer, E.: "Co-existing Lobar Adenocarcinoma and Cystic Disease of Lung," Ann. Int. Mea., 34:498, 1951. 2 Graham, E. A.: Discussion on "Surgical Thoracic Tumors in Navy Personnel" by W. L. Watson and Henry D. Diamond, and "Pulmonary Cysts" by Herman J . Moersch and O. Theron Clagett, J . Thor. Surg., 16:194,1947. 3 Graham, E . A., Singer, J. J ., and Ballon, H. C. : Surgical Diseases of the Chest, Philadelphia, Lea and Feblger, 1935. 4 Korol, E.: "Observation on Cystic and Bullous Emphysema of the Lungs, A study of 100 Cases," Dis. Chest, 13:669, 1947. 5 Korol, E.: "The Correlation of Carcinoma and Congenital Cystic Emphysema of the Lungs: Report of Ten Cases," Dis. Chest, 23:403, 1953. 6 Larkin, J. C., Jr., and Phillips, S .: "Carcinoma Complicating Cyst of Lung," Dis. Chest, 27:453. 1955. 7 McKusick, V. A., and Fisher, A. M.: "Congenital Cystic Disease of the Lung with Progressive Pulmonary Fibrosis and Carcinomatosis," Ann. Int. Mea., 48:774, 1958. 8 Moersch, H . J., and Clagett, O . T.: "Pulmonary Cysts," J. Thor. Surg., 16:179, 1947. 9 Murphy, J. D ., and Piver, J . D. : "Cystic Disease of the Lung," Dis. Chest, 19:454, 1951.
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10 Peabody, J. W., Katz, S., and Davis, E. W.: "Bronchial Carcinoma Arising ina Lung Cyst," Am. J. Roentgenol., 77:1048,1957. 11 Peabody, J. W., Rupnik, E. J., and Hammer, J. M.: "Bronchial Carcinoma Masquerading as A Thin Walled Cyst," Am. J. RoentgenoZ., 77:1051, 1957. 12 Rogers, W. L.: "Cystic Disease of the Lung," West. J. Surg., 56:157, 1948. 13 West, J. P., and Van SChoonhoven, P. V.: "Carcinoma of the Lung Developing in a Congenital Cyst," swaer», 42:1071, 1957. 14 Womack, N. A., and Graham, E. A.: "Epithelial Metaplasia in Congenital Cystic Disease of the Lung; Its Possible Relation to Carcinoma of the Bronchus," Am. J. Path., 17:645, 1941. 15 Womack, N. A., and Graham, E. A.: "Developmental Abnormalities of the Lung and Bronchogenic Carcinoma," Arch. Path., 34:301,1942.
OPISTHORCHIASIS WITH PULMONARY INVOLVEMENT The diagnosis of opisthorchiasis In this case was establlshed by the repeated finding of the ova In the feces. The pulmonary lesion In the right lower lung field. with greenish sputum which showed a positive bUe test, suggested a connection with the biliary tract. A/ca/igeneJ laeca/iJ found In the sputum was Interpreted as a secondary Invader. The Introduetlon of radiopaque substance Into the liver showed finger-like dilatations of the biliary tract. This Is typical of advanced cases of opisthorchiasis. The radiopaque substance was also found In the lower lung field. suggesting the presence of a connection between the liver and the lesion In the lung. This was confirmed at necropsy. A thorough search of the available literature falls to disclose any previously reported case of Intrathoracic opisthorchiasis diagnosed ante-mortem. Prljyanonda. B.. and Tandhanand. S.: "Opisthorchiasis with Pulmonary Involvement." Ann. Int. MeJ., 54 :795. 1961.
COMPARATIVE STUDIES OF PROTOVERATRINE A AND
PROTOVERATRINE B INTRAVENOUSLY IN HYPERTENSIVE MAN Comparative studies of Intravenously administered protoveratrine A and protoveratrine B In hypertensive man Indicate that the alkaloids have qualitatively similar hypotensive. bradycrotic and emetic actions. but quantitatively different hypotensive and emetic potency. Protoveratrine B was better tolerated than protoveratrtne A. Indeed. protoveratrine B had little emetic activity when given Intravenously In doses producing nomotenslon. Protoveratrine B appears to have properties that warrant Its preferential selection for parenteral administration. The strong hypotensive activity of intravenously administered protoveratrine B, Its development of maximal action within minutes, and Its pulse-slowing effect favor Its use In those hypertensive states In which almost Immediate lowering of the blood pressure Is Indicated. Winer, B. M.: ·'Comparattve Studies of Protoveratrine A and Protoveratrlne B Intravenously In Hypertensive Man." Circllia/ion. 22: 1074, 1960.
In 1941, Womack and Graham" first called attention to the occurrence of squamous metaplasia in the lining of congenital lung cysts. This observation was based upon the study of three cases, which they felt suggested a pathogenetic role for this change in the formation of squamous cell carcinoma. Both prior to and since this article by Womack and Graham, several papersr":":"... have appeared which either cite or report cases of carcinoma associated with lung cysts. However, most of these 1·...... have been cases of independent bronchial carcinoma in a cystic lung; or they have been so poorly documented that no direct relationship was apparent between a carcinoma and a pulmonary cyst. If one accepts the criticism offered by some authors," there are only three cases':":" in the English literature in which there is irrefutable documentation of a carcinoma arising in the wall of a congenital pulmonary cyst. The following case is presented as another example of this occurrence.
Case Report V.RL.: This 30 year-old white man cigarette smoker was known to have a solitary right lower lobe cyst, which had. been detected on a routine physical examination one year prior to admission (Fig. 1). He had received no treatment and had. been asvmp-
FIGURE 1: Chest roentgenogram taken one year prior to admission showing solitary pulmonary cyst in right lower lobe . -From the Department of Pathology, U. S . Naval Hospital. - -The views expressed are those of the author anJ do not necessarily represent the views of the Bureau of Medicine and Surgery, Navy Department. tPresently at the Beth Israel HosPital, New York City. New York. 552
Vol. 40
CARCINOMA IN CONGENITAL LUNG CYST
553
tomatic until one week before his admission to the U. S. Naval Hospital, when he developed an aching pain in the right posterior aspect of the chest. The physical findings were eSBent1ally normal except for tenderness over the posterior portion of the right sixth through eighth ribs. Chest roentgenograms revealed a single cyst in the superior portion of the right lower lobe. Comparison with the previous film (Fig. 1) showed no change in the size or configuration of the Cyst. Right thoracotomy was performed, at which time the lower lobe was found to be adherent to the seventh rib posteriorly. A frozen section of the lung revealed an oat cell carcinoma. Right lower lobectomy was performed. The surgical specimen disclosed a thin walled spherical cyst located between the posterior basal and lateral basal segments. The cyst measured 8.0 em. In diameter. On the postero-lateral wall of the cyst there was a firm greyish-white tumor nodule which measured 1.0 em. in greatest diameter (Fig. 2) . The tumor 1n1Utrated the adjacent lung, where it had been separated from the rib cage . There were four small communications between the cyst and the bronchial tree ; however, there was no bronchus related to the tumor. All of the major bronchi of the lobe were unremarkable. Microscopic sections revealed the cyst to be lined with typical bronchial epithelium (Fig. 3). The cyst wall was composed of a thin layer of chronically infiamed fibrous tissue and rare smooth muscle elements. The tumor. which arose from the lining of the cyst, was composed of well differentiated squamous cell carcinoma (Fig. 4) mixed with areas of oat cell carcinoma. The tumor inft1trated the surrounding lung tissue and pleura. A hllar lymph node contained metastatic squamous cell carcinoma. The immediate postoperative course was uncomplicated. Because of the local rib invasion and hUar node spread, x-ray and nitrogen mustard therapy were instituted. His condition deteriorated progressively and he expired seven months after admission. Postmortem examination revealed a large tumor mass in the right hemithorax. The tumor 1n1Utrated the third through the eighth vertebral bodies and the adjoining ribs and soft tissues. There were distant metastases in the hilar lymph nodes, both adrenal glands and liver. On microscopic study all of the tumor tissue was found to be composed of poorly differentiated squamous cell carcinoma.
Comment This case bears out the contention of Womack and Graham lS and Graham" that the bronchial epithelium of congenital lung cysts can give rise to squamous cell carcinoma. Perhaps the development of carcinoma should be considered as a PQ881ble complication when planning the management of a case of congenital lung cyst. This consideration might serve as another indication for surgical intervention in these cases.·
FIGURE 2: Lung cyst showing squamous cell carcinoma (arrow) arising in the wall.
554
LT. STANLEY BAUER
Nov. . 1961
Figure 3
Figure 4
FIGURE 3: Section through cyst wall showing typical bronchial epithelium. (H . and E. DOO) . FIGURE 4: Section through tumor mass showing welld1fferentiated squamous cell carcinoma. (H . and E. DOO) . REFERENCES
1 Bass, H. E ., and Singer, E.: "Co-existing Lobar Adenocarcinoma and Cystic Disease of Lung," Ann. Int. Mea., 34:498, 1951. 2 Graham, E. A.: Discussion on "Surgical Thoracic Tumors in Navy Personnel" by W. L. Watson and Henry D. Diamond, and "Pulmonary Cysts" by Herman J . Moersch and O. Theron Clagett, J . Thor. Surg., 16:194,1947. 3 Graham, E . A., Singer, J. J ., and Ballon, H. C. : Surgical Diseases of the Chest, Philadelphia, Lea and Feblger, 1935. 4 Korol, E.: "Observation on Cystic and Bullous Emphysema of the Lungs, A study of 100 Cases," Dis. Chest, 13:669, 1947. 5 Korol, E.: "The Correlation of Carcinoma and Congenital Cystic Emphysema of the Lungs: Report of Ten Cases," Dis. Chest, 23:403, 1953. 6 Larkin, J. C., Jr., and Phillips, S .: "Carcinoma Complicating Cyst of Lung," Dis. Chest, 27:453. 1955. 7 McKusick, V. A., and Fisher, A. M.: "Congenital Cystic Disease of the Lung with Progressive Pulmonary Fibrosis and Carcinomatosis," Ann. Int. Mea., 48:774, 1958. 8 Moersch, H . J., and Clagett, O . T.: "Pulmonary Cysts," J. Thor. Surg., 16:179, 1947. 9 Murphy, J. D ., and Piver, J . D. : "Cystic Disease of the Lung," Dis. Chest, 19:454, 1951.
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10 Peabody, J. W., Katz, S., and Davis, E. W.: "Bronchial Carcinoma Arising ina Lung Cyst," Am. J. Roentgenol., 77:1048,1957. 11 Peabody, J. W., Rupnik, E. J., and Hammer, J. M.: "Bronchial Carcinoma Masquerading as A Thin Walled Cyst," Am. J. RoentgenoZ., 77:1051, 1957. 12 Rogers, W. L.: "Cystic Disease of the Lung," West. J. Surg., 56:157, 1948. 13 West, J. P., and Van SChoonhoven, P. V.: "Carcinoma of the Lung Developing in a Congenital Cyst," swaer», 42:1071, 1957. 14 Womack, N. A., and Graham, E. A.: "Epithelial Metaplasia in Congenital Cystic Disease of the Lung; Its Possible Relation to Carcinoma of the Bronchus," Am. J. Path., 17:645, 1941. 15 Womack, N. A., and Graham, E. A.: "Developmental Abnormalities of the Lung and Bronchogenic Carcinoma," Arch. Path., 34:301,1942.
OPISTHORCHIASIS WITH PULMONARY INVOLVEMENT The diagnosis of opisthorchiasis In this case was establlshed by the repeated finding of the ova In the feces. The pulmonary lesion In the right lower lung field. with greenish sputum which showed a positive bUe test, suggested a connection with the biliary tract. A/ca/igeneJ laeca/iJ found In the sputum was Interpreted as a secondary Invader. The Introduetlon of radiopaque substance Into the liver showed finger-like dilatations of the biliary tract. This Is typical of advanced cases of opisthorchiasis. The radiopaque substance was also found In the lower lung field. suggesting the presence of a connection between the liver and the lesion In the lung. This was confirmed at necropsy. A thorough search of the available literature falls to disclose any previously reported case of Intrathoracic opisthorchiasis diagnosed ante-mortem. Prljyanonda. B.. and Tandhanand. S.: "Opisthorchiasis with Pulmonary Involvement." Ann. Int. MeJ., 54 :795. 1961.
COMPARATIVE STUDIES OF PROTOVERATRINE A AND
PROTOVERATRINE B INTRAVENOUSLY IN HYPERTENSIVE MAN Comparative studies of Intravenously administered protoveratrine A and protoveratrine B In hypertensive man Indicate that the alkaloids have qualitatively similar hypotensive. bradycrotic and emetic actions. but quantitatively different hypotensive and emetic potency. Protoveratrine B was better tolerated than protoveratrtne A. Indeed. protoveratrine B had little emetic activity when given Intravenously In doses producing nomotenslon. Protoveratrine B appears to have properties that warrant Its preferential selection for parenteral administration. The strong hypotensive activity of intravenously administered protoveratrine B, Its development of maximal action within minutes, and Its pulse-slowing effect favor Its use In those hypertensive states In which almost Immediate lowering of the blood pressure Is Indicated. Winer, B. M.: ·'Comparattve Studies of Protoveratrine A and Protoveratrlne B Intravenously In Hypertensive Man." Circllia/ion. 22: 1074, 1960.