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Congenital bronchiolar cysts simulating intralobar sequestration
Congenital Bronchiolar Intralobar By MICHEL
Cysts Simulating
Sequestration
S. SLIM, PHILIP SAHYOUN AND JIRAYR BALIKIAN
concerns a case of congenital bronchiolar cysts simulating sequestration in a 9 month old infant. These cysts were in direct communication with the main bronchus, and had features reminiscent of sequestration. Because of the rarity of this condition in infancy, and the unusual pathological features presented by this patient, we were prompted to prepare this communication. HIS REPORT
T intralobar
CASE HISTORY A 9 month old Lebanese boy was admitted to the Hospital of the American University of Beirut in January, 1966, with the history of persistent cough and mild difficulty in breathing of 3 weeks’ duration. The mother reported that during the first few weeks of life the baby occasionally choked and became cyanotic after feedings. At the age of 4 months, he developed a severe respiratory infection which responded to antibiotics and cough medicine. He was then well until 3 weeks prior to admission when cough and fever recurred. On physical examination, he was well developed and was not cyanotic. His weight was 7.5 Kg. and his temperature was 37.5” C. rectally. Breath sounds were absent and dullness was elicited on percussion over the right lower lobe area. No rales were heard. 21,350W.B.C., and on diEerentia1 smear Blood count showed 38 per cent hematocrit; 40 per cent neutrophils, 57 per cent lymphocytes, 2 per cent monocytes and 1 per cent eosinophiles. Chest roentgenogram (Fig. 1A) and fluoroscopy showed elevation and limited motion of the right side of the diaphragm. No shift of the mediastinum was observed during respiration. The esophagogram did not show a fistulous communication between the esophagus Bronchography, using 15 and the tracheobronchial tree. ml. of aqueous Dionosil,@ revealed a large, smooth walled cavity in the posterior part of the right base communicating with the bronchus (Fig. lB, C.). The bronchi in the right lower lobe were dilated. These findings suggested the diagnosis of sequestration of the lung. Following this study, the patient’s temperature rose to 39.8” C. and was not accompanied by a change in the leucocyte count. He was discharged from the hospital and was re-admitted for thoracotomy 3 months later. The mother stated that the patient stopped coughing about a month before the thoracotomy. Chest roentgenogram one day before operation showed residual opaque material in the cavity at the right base (Fig. 1D). A right lower lobectomy was performed in April. 1966. At operation, the right lower lobe was collapsed and adherent to the diaphragm and chest wall. The posterior basal segment was attached to the diaphragm by a band containing an anomalous vessel. During dissection of the lobe. the cyst was opened and viscid yellowish gray material drained. No growth was obtained on culture of this material. These findings were reminiscent of intralobar sequestration. On section, the main cyst was connected with the parent bronchus From the Departments of Surgery, Pathology and Radiology, School of Medicine, American University of Beirut, Beirut, Lebanon. MICHEL S. SLIM, M.D.: Assistant Professor of Pediatric Surgery, American University of Beirut. PHILIP SAHYOUN, M.D.: Professor of Pathology, American University of Beirut. JIRAYRBALIKIAN, M.D.: Assistant Professor of Radiology, American University of Beirut. 60
JOURNAL
OF
PEDIATRIC SURGERY, VOL. 3, No. l,lP~. I (FEB.). 1968
Fig. 1.-A, Chest roentgenogram 3 months before operation reveals an elevated right diaphragm. B and C, Bronchographv reveals a communicating cyst in the posterior basal segment of the right lower lobe and dilatation of the adjacent bronchi. D and E, Films taken one day and 3 months after bronchographv respectivelv shorn retention of aqueous DionosiI@ in the intralobar cvst. by a bronchiole having a narrow lumen. hlicroscopically, the cyst wall was lined by fragments of degenerating bronchial epithelium and by granulation tissue containing numerous foreign body giant cells some of which were vacuolated (Fig. 2A). Alcian blue stain for mucin counterstained with neutral red showed a blue-staining material in the bronchial gland spaces of the cyst wall and a second material staining with neutral red which was incorporated in the giant cells. The lumen of the cyst contained a crystalline material mixed with mucin (Fig. 2B). The outer layer of the cyst wall consisted of collagenous fibrous tissue. Minimal leucocytic infiltration by lymphoid cells was seen in the surrounding lung parenchyma. The vessels showed no changes in their walls. The bronchioles in the right lower lobe showed a similar reaction to that in the wall of the main cyst. The peribronchial mucous glands showed the same type of degenerative changes as the mucous glands in the wall of the cyst. The histological findings suggested that the cyst was a congenital tnalformation rather than the result of an infection. His postoperative course was uneventful and he was discharged in good condition one week after surgery. He was well when last examined 1 month after operation.
Congenital ized until
pulmonary
the intracavitary
cysts
are rare
pressure
in infancy.‘.”
increases,
They
compromising
remain
unrecog-
the function
of
the surrounding structures, or infection sets in, causing a protracted febrile course and resulting in hemoptysis. Intralobar sequestration does not usually become manifest clinically in the first year of life.7 On review of the English literature, we were able to find three case reports of infants treated for intralobar sequestration in the first year of life.“~“’ The cysts which were seen in
62
SLIM
ET
AL.
Fig. 2. -A, Photomicrograph of the resected lobe showing wall of the main cyst with destruction of mucosa and mucous glands and inclusion of mucus in giant cells (Alcian blue stain, 50 x magnifkation) . B, Photomicrograph of the resected lobe showing contents of the main cyst: mucus surrounded by crystalline material ( Alcian blue stain, 50 x magnification).
63
CONGENITALBRONCHIOLAR CYSTS the affected lobes of these three infants did not communicate
with the main
bronchial tree. We have elected to describe the findings in our case as bronchiolar cysts simulating intralobar sequestration on basis of the morphological features. The location of the cyst in the lower lobe, the posteromedial direction of the long axis of the main cyst, the nature of the cyst contents and the operative finding of an anomalous artery crossing the dome of the diaphragm are reminiscent of intralobar sequestration. However, the presence of a fairly goodsized commmlication between the main cyst and the right main stem bronchus does not conform with other reports on children with intralobar sequestration.“~“~F-3” Despite this communication our patient presented evidence of unusually prolonged retention of aqueous Dionosil for 3 months after its instillation . We do not believe the cysts reported here to be primarily on an infectious basis.l The previously described pathologic and operative data and the course of the infant’s illness favor a congenital origin with infection as a secondarv event. Aortography has been recommended for the preoperative confirmation of sequestration :10but is not necessary if the surgeon is careful to divide and ligate all major adhesions between clamps. These adhesions envelop densely the anomalous vessels to the involved part of lung. A preoperative aortogram in our case would have defined the origin of the anomalous vessel but would not have changed the nature of the surgical procedure. We agree with others that lobectomy is the procedure of choice for intralobar sequestration and complicated congenital pulmonary cysts in patients with adequate pulmonary reserve. lG The timing and the extent of resection should be decided upon according to distribution of the lesion, to the presence or absence of superimposed infection and to the pulmonary function of the patient. SUMMARY In a case of bronchiolar cysts simulating intralobar sequestration in a 9 month old infant, the main cyst occupied the right posterior basal segment. was attached to the diaphragm by a band containing an anomalous vessel, but had an unusual communication with the lower lobe bronchus. The history of the patient was characterized by repeated respiratory infections. The treatment consisted of a right lower lobectomy. The rarity of this malformation in infants under the age of 1 year is evident on review of the literature. SUMMARIO
IN INTERLINGUA
Es presentate le case de un infante de 9 menses de etate in qui cystes bronchiolar simulava sequestration intralobar. Le cyste principal occupava le segment0 basal dextero-posterior. 1110 esseva attachate al diaphragma per un ligament0 continente un vaso anormal e habeva 1m communication inusual con le broncho de1 lobo inferior. Le historia clinic de1 patiente esseva characterkate oer repetite infectiones respiratori. Le tractamento consisteva de lobectomia dextero-inferior. Un revista de1 litteratura demonstra le raritate de iste malformation in infantry de minus qlw nn anno de etate.
SLIM
64
ET
AL.
REFERENCES 1. Caffey, J.: On the natural regression of pulmonary cysts during early infancy. Pediatrics 11:48, 1953. 2. Lederer, M., quoted by Schenck, S. G.: Congenital cystic disease of the lung. Amer. J. Roentgen. 35:604, 1936. 3. Ravitch, M. M., and Hardy, J. B.: Congenital cystic disease of the lung in infants and in children. Arch. Surg. 59:1, 1949. 4. Brunner, S., Poulsen, P. T., and Vesterdal, J.: Cysts of the lung in infants and children. Acta Pediat. 49:39, 1960. 5. Slim. M. S., and Melhem, It. E.: Congenital pulmonary air cysts. Arch. Surg. 88: 923, 1964. 6. Swan, H., and Aragon, C. E.: Surgical treatment of pulmonary cysts in infancy. Pediatrics 14:651, 1954. 7. Potter, E. L.: Pathology of the Fetus and the Infant (2nd ed.). Yearbook Medical Publishers, Chicago, Ill., 1962. 8. Deaton, W. R., and Smith, R. M.: Sequestration of the left lower lobe of the lung. Arch. Surg. 74:149, 1957. 9. Mannix, E. P., and Haight, C.: Anomalous pulmonary arteries and cystic disease of the lung. Medicine 34:193, 1955. 10. Talelak, P.: Pulmonary sequestration. Arch. Dis. Child. 35:57, 1960. 11. Abdel, K. P.: Intralobar sequestration of the lung. Proc. Royal Sot. Med. 54:691, 1961. 12. Anderson, A. E., Brown, J. B., and Roesch, C. B.: Aberrant pulmonary artery with intralobar sequestration. Ann. Int. Med. 46: 1092, 1957. 13. Asp, K., Pasila, M., and Sulamaa, M.: Diagnosis and treatment of pulmonary sequestration. Acta Chir. Stand. 127:303, 1964. 14. Beskin, C. A.: Intralobar enteric sequestration of the lung containing aberrant pancreas. J. Thor. Surg. 41:314, 1961. 15. Borric, J., Lichter, I.. and Rodda, R. A.: Intralobar pulmonary sequestration. Brit. J. Surg. 50:623, 1962-63. 16. Boyd, C.: Intralobar pulmonary sequestration. Dis. Chest 24:162, 1953. 17. Claman, M. A., and Ehrenhaft, J. L.: Bronchopulmonary sequestration. J. Thor. Surg. 39:531, 1960. 18. Cole, F. H., Alley, F. H., and Jones, R. S.: Aberrant systemic arteries the lower lung. Surg. Gynec. Obstet. 93:589, 1951.
19. Cooley, J. C.: Intralobar bronchopulmonary sequestration. Dis. Chest 42:95, 1962. 20. Douglass, R.: Anomalous pulmonary vessels. J. Thor. Surg. 17:712, 1948. 21. Eaker, A. B., Hannon, J. L., and French, S. W.: Pulmonary sequestration. Amer. J. Surg. 95:31, 1958. 22. Gerard, F. P., and Lyons, H. A.: Anomalous artery in intralobar bronchopulmonary sequestration. New Eng. J. Med. 259:662, 1958. 23. Hardin, C. A.: Intralobar bronchopulmonary sequestration of the lung. Dis. Chest. 38:202, 1960. 24. Harris, H. A., and Lewis, I.: Anomalies of the lungs with special reference to the danger of abnormal vessels in lobectomy. J. Thor. Surg. 9:666, 1940. 25. Kafka, V., and Beco, V.: Simultaneous intraand extra-pulmonary sequestration. Arch. Dis. Child. 35:51, 1960. 26. Lalli, A., Carlson, R., and Adams, W. E.: Intralobar pulmonary sequestration. Arch. Surg. 69:797, 1954. 27. McDowell, C., Robb, D., Hinds, J. R., and Nicks, R.: A case on intralobar sequestration of the lung associated with an abnormal pulmonary artery. Brit. J. Surg. 39:87, 1951-52. 28. Pryce, D. M.: Lower accessory pulmonary artery with intralobar sequestration of lung: a report of seven cases. J. Path. Bact. 58:457, 1946. 29. Simopoulos, A. P., Rosenblum, D. J., Mazumdar, H., and Kiely, B.: Intralobar bronchopulmonary sequestration in children. J. Dis. Child. 97:796, 1959. 30. Turk. L. N., and Lindskog, C. E.: The importance of angiographic diagnosis in intralobar pulmonary sequestration. J. Thor. Surg. 41:299, 1961. 31. Warner, C. L., Britt, R. L., and Riley, H. D.: Broncho-pulmonary sequestration in infancy and childhood. J. Ped. 53:521, 1958. 32. Witten, D. M., Clagett, 0. R., and Woolner, L. W.: Intralobar bronchopulmonary sequestration involving the upper lobes. J. Thor. Surg. 43:523, 1962. 33. Wyman, S. M., and Eyler, W. R.: Anomalous pulmonary artery from the aorta associated with intrapulmonary cysts (intralobar sequestration of lung). Radiology 50: 658, 1952.
Cysts Simulating
Sequestration
S. SLIM, PHILIP SAHYOUN AND JIRAYR BALIKIAN
concerns a case of congenital bronchiolar cysts simulating sequestration in a 9 month old infant. These cysts were in direct communication with the main bronchus, and had features reminiscent of sequestration. Because of the rarity of this condition in infancy, and the unusual pathological features presented by this patient, we were prompted to prepare this communication. HIS REPORT
T intralobar
CASE HISTORY A 9 month old Lebanese boy was admitted to the Hospital of the American University of Beirut in January, 1966, with the history of persistent cough and mild difficulty in breathing of 3 weeks’ duration. The mother reported that during the first few weeks of life the baby occasionally choked and became cyanotic after feedings. At the age of 4 months, he developed a severe respiratory infection which responded to antibiotics and cough medicine. He was then well until 3 weeks prior to admission when cough and fever recurred. On physical examination, he was well developed and was not cyanotic. His weight was 7.5 Kg. and his temperature was 37.5” C. rectally. Breath sounds were absent and dullness was elicited on percussion over the right lower lobe area. No rales were heard. 21,350W.B.C., and on diEerentia1 smear Blood count showed 38 per cent hematocrit; 40 per cent neutrophils, 57 per cent lymphocytes, 2 per cent monocytes and 1 per cent eosinophiles. Chest roentgenogram (Fig. 1A) and fluoroscopy showed elevation and limited motion of the right side of the diaphragm. No shift of the mediastinum was observed during respiration. The esophagogram did not show a fistulous communication between the esophagus Bronchography, using 15 and the tracheobronchial tree. ml. of aqueous Dionosil,@ revealed a large, smooth walled cavity in the posterior part of the right base communicating with the bronchus (Fig. lB, C.). The bronchi in the right lower lobe were dilated. These findings suggested the diagnosis of sequestration of the lung. Following this study, the patient’s temperature rose to 39.8” C. and was not accompanied by a change in the leucocyte count. He was discharged from the hospital and was re-admitted for thoracotomy 3 months later. The mother stated that the patient stopped coughing about a month before the thoracotomy. Chest roentgenogram one day before operation showed residual opaque material in the cavity at the right base (Fig. 1D). A right lower lobectomy was performed in April. 1966. At operation, the right lower lobe was collapsed and adherent to the diaphragm and chest wall. The posterior basal segment was attached to the diaphragm by a band containing an anomalous vessel. During dissection of the lobe. the cyst was opened and viscid yellowish gray material drained. No growth was obtained on culture of this material. These findings were reminiscent of intralobar sequestration. On section, the main cyst was connected with the parent bronchus From the Departments of Surgery, Pathology and Radiology, School of Medicine, American University of Beirut, Beirut, Lebanon. MICHEL S. SLIM, M.D.: Assistant Professor of Pediatric Surgery, American University of Beirut. PHILIP SAHYOUN, M.D.: Professor of Pathology, American University of Beirut. JIRAYRBALIKIAN, M.D.: Assistant Professor of Radiology, American University of Beirut. 60
JOURNAL
OF
PEDIATRIC SURGERY, VOL. 3, No. l,lP~. I (FEB.). 1968
Fig. 1.-A, Chest roentgenogram 3 months before operation reveals an elevated right diaphragm. B and C, Bronchographv reveals a communicating cyst in the posterior basal segment of the right lower lobe and dilatation of the adjacent bronchi. D and E, Films taken one day and 3 months after bronchographv respectivelv shorn retention of aqueous DionosiI@ in the intralobar cvst. by a bronchiole having a narrow lumen. hlicroscopically, the cyst wall was lined by fragments of degenerating bronchial epithelium and by granulation tissue containing numerous foreign body giant cells some of which were vacuolated (Fig. 2A). Alcian blue stain for mucin counterstained with neutral red showed a blue-staining material in the bronchial gland spaces of the cyst wall and a second material staining with neutral red which was incorporated in the giant cells. The lumen of the cyst contained a crystalline material mixed with mucin (Fig. 2B). The outer layer of the cyst wall consisted of collagenous fibrous tissue. Minimal leucocytic infiltration by lymphoid cells was seen in the surrounding lung parenchyma. The vessels showed no changes in their walls. The bronchioles in the right lower lobe showed a similar reaction to that in the wall of the main cyst. The peribronchial mucous glands showed the same type of degenerative changes as the mucous glands in the wall of the cyst. The histological findings suggested that the cyst was a congenital tnalformation rather than the result of an infection. His postoperative course was uneventful and he was discharged in good condition one week after surgery. He was well when last examined 1 month after operation.
Congenital ized until
pulmonary
the intracavitary
cysts
are rare
pressure
in infancy.‘.”
increases,
They
compromising
remain
unrecog-
the function
of
the surrounding structures, or infection sets in, causing a protracted febrile course and resulting in hemoptysis. Intralobar sequestration does not usually become manifest clinically in the first year of life.7 On review of the English literature, we were able to find three case reports of infants treated for intralobar sequestration in the first year of life.“~“’ The cysts which were seen in
62
SLIM
ET
AL.
Fig. 2. -A, Photomicrograph of the resected lobe showing wall of the main cyst with destruction of mucosa and mucous glands and inclusion of mucus in giant cells (Alcian blue stain, 50 x magnifkation) . B, Photomicrograph of the resected lobe showing contents of the main cyst: mucus surrounded by crystalline material ( Alcian blue stain, 50 x magnification).
63
CONGENITALBRONCHIOLAR CYSTS the affected lobes of these three infants did not communicate
with the main
bronchial tree. We have elected to describe the findings in our case as bronchiolar cysts simulating intralobar sequestration on basis of the morphological features. The location of the cyst in the lower lobe, the posteromedial direction of the long axis of the main cyst, the nature of the cyst contents and the operative finding of an anomalous artery crossing the dome of the diaphragm are reminiscent of intralobar sequestration. However, the presence of a fairly goodsized commmlication between the main cyst and the right main stem bronchus does not conform with other reports on children with intralobar sequestration.“~“~F-3” Despite this communication our patient presented evidence of unusually prolonged retention of aqueous Dionosil for 3 months after its instillation . We do not believe the cysts reported here to be primarily on an infectious basis.l The previously described pathologic and operative data and the course of the infant’s illness favor a congenital origin with infection as a secondarv event. Aortography has been recommended for the preoperative confirmation of sequestration :10but is not necessary if the surgeon is careful to divide and ligate all major adhesions between clamps. These adhesions envelop densely the anomalous vessels to the involved part of lung. A preoperative aortogram in our case would have defined the origin of the anomalous vessel but would not have changed the nature of the surgical procedure. We agree with others that lobectomy is the procedure of choice for intralobar sequestration and complicated congenital pulmonary cysts in patients with adequate pulmonary reserve. lG The timing and the extent of resection should be decided upon according to distribution of the lesion, to the presence or absence of superimposed infection and to the pulmonary function of the patient. SUMMARY In a case of bronchiolar cysts simulating intralobar sequestration in a 9 month old infant, the main cyst occupied the right posterior basal segment. was attached to the diaphragm by a band containing an anomalous vessel, but had an unusual communication with the lower lobe bronchus. The history of the patient was characterized by repeated respiratory infections. The treatment consisted of a right lower lobectomy. The rarity of this malformation in infants under the age of 1 year is evident on review of the literature. SUMMARIO
IN INTERLINGUA
Es presentate le case de un infante de 9 menses de etate in qui cystes bronchiolar simulava sequestration intralobar. Le cyste principal occupava le segment0 basal dextero-posterior. 1110 esseva attachate al diaphragma per un ligament0 continente un vaso anormal e habeva 1m communication inusual con le broncho de1 lobo inferior. Le historia clinic de1 patiente esseva characterkate oer repetite infectiones respiratori. Le tractamento consisteva de lobectomia dextero-inferior. Un revista de1 litteratura demonstra le raritate de iste malformation in infantry de minus qlw nn anno de etate.
SLIM
64
ET
AL.
REFERENCES 1. Caffey, J.: On the natural regression of pulmonary cysts during early infancy. Pediatrics 11:48, 1953. 2. Lederer, M., quoted by Schenck, S. G.: Congenital cystic disease of the lung. Amer. J. Roentgen. 35:604, 1936. 3. Ravitch, M. M., and Hardy, J. B.: Congenital cystic disease of the lung in infants and in children. Arch. Surg. 59:1, 1949. 4. Brunner, S., Poulsen, P. T., and Vesterdal, J.: Cysts of the lung in infants and children. Acta Pediat. 49:39, 1960. 5. Slim. M. S., and Melhem, It. E.: Congenital pulmonary air cysts. Arch. Surg. 88: 923, 1964. 6. Swan, H., and Aragon, C. E.: Surgical treatment of pulmonary cysts in infancy. Pediatrics 14:651, 1954. 7. Potter, E. L.: Pathology of the Fetus and the Infant (2nd ed.). Yearbook Medical Publishers, Chicago, Ill., 1962. 8. Deaton, W. R., and Smith, R. M.: Sequestration of the left lower lobe of the lung. Arch. Surg. 74:149, 1957. 9. Mannix, E. P., and Haight, C.: Anomalous pulmonary arteries and cystic disease of the lung. Medicine 34:193, 1955. 10. Talelak, P.: Pulmonary sequestration. Arch. Dis. Child. 35:57, 1960. 11. Abdel, K. P.: Intralobar sequestration of the lung. Proc. Royal Sot. Med. 54:691, 1961. 12. Anderson, A. E., Brown, J. B., and Roesch, C. B.: Aberrant pulmonary artery with intralobar sequestration. Ann. Int. Med. 46: 1092, 1957. 13. Asp, K., Pasila, M., and Sulamaa, M.: Diagnosis and treatment of pulmonary sequestration. Acta Chir. Stand. 127:303, 1964. 14. Beskin, C. A.: Intralobar enteric sequestration of the lung containing aberrant pancreas. J. Thor. Surg. 41:314, 1961. 15. Borric, J., Lichter, I.. and Rodda, R. A.: Intralobar pulmonary sequestration. Brit. J. Surg. 50:623, 1962-63. 16. Boyd, C.: Intralobar pulmonary sequestration. Dis. Chest 24:162, 1953. 17. Claman, M. A., and Ehrenhaft, J. L.: Bronchopulmonary sequestration. J. Thor. Surg. 39:531, 1960. 18. Cole, F. H., Alley, F. H., and Jones, R. S.: Aberrant systemic arteries the lower lung. Surg. Gynec. Obstet. 93:589, 1951.
19. Cooley, J. C.: Intralobar bronchopulmonary sequestration. Dis. Chest 42:95, 1962. 20. Douglass, R.: Anomalous pulmonary vessels. J. Thor. Surg. 17:712, 1948. 21. Eaker, A. B., Hannon, J. L., and French, S. W.: Pulmonary sequestration. Amer. J. Surg. 95:31, 1958. 22. Gerard, F. P., and Lyons, H. A.: Anomalous artery in intralobar bronchopulmonary sequestration. New Eng. J. Med. 259:662, 1958. 23. Hardin, C. A.: Intralobar bronchopulmonary sequestration of the lung. Dis. Chest. 38:202, 1960. 24. Harris, H. A., and Lewis, I.: Anomalies of the lungs with special reference to the danger of abnormal vessels in lobectomy. J. Thor. Surg. 9:666, 1940. 25. Kafka, V., and Beco, V.: Simultaneous intraand extra-pulmonary sequestration. Arch. Dis. Child. 35:51, 1960. 26. Lalli, A., Carlson, R., and Adams, W. E.: Intralobar pulmonary sequestration. Arch. Surg. 69:797, 1954. 27. McDowell, C., Robb, D., Hinds, J. R., and Nicks, R.: A case on intralobar sequestration of the lung associated with an abnormal pulmonary artery. Brit. J. Surg. 39:87, 1951-52. 28. Pryce, D. M.: Lower accessory pulmonary artery with intralobar sequestration of lung: a report of seven cases. J. Path. Bact. 58:457, 1946. 29. Simopoulos, A. P., Rosenblum, D. J., Mazumdar, H., and Kiely, B.: Intralobar bronchopulmonary sequestration in children. J. Dis. Child. 97:796, 1959. 30. Turk. L. N., and Lindskog, C. E.: The importance of angiographic diagnosis in intralobar pulmonary sequestration. J. Thor. Surg. 41:299, 1961. 31. Warner, C. L., Britt, R. L., and Riley, H. D.: Broncho-pulmonary sequestration in infancy and childhood. J. Ped. 53:521, 1958. 32. Witten, D. M., Clagett, 0. R., and Woolner, L. W.: Intralobar bronchopulmonary sequestration involving the upper lobes. J. Thor. Surg. 43:523, 1962. 33. Wyman, S. M., and Eyler, W. R.: Anomalous pulmonary artery from the aorta associated with intrapulmonary cysts (intralobar sequestration of lung). Radiology 50: 658, 1952.