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Malignant fibrous histiocytoma of left atrium
The Journal of Thoracic and Cardiovascular Surgery
4 5 0 Brief communications
Table I. Chylopericardium after CABG operation Patient (age. sex)
Operation
Cause
Pollard et al.' (1981)
57, M
CABGx4
Schiessler et al." (1984) Bakayet al.' (1984)
45, M 41, M
CABGX I CABGX3
Yes Yes
Lee et al. (1987)
49, M
CABGX
No
Left subclavian line Not known Left jugular line Not known
Authors
I
Treatment
Drainage, diet Drainage, diet Subxiphoid pericardioperitoneal fistula Drainage, diet, ligation thoracic duct, pleuroperitoneal shunt
Legend: CABG, Coronary artery bypass graft. CPB, Cardiopulmonary bypass.
fluid accumulation. The follow-up chest film taken 1 year later showed no abnormalities.
Discussion. Azizkhan," Milsom,' and their associates reported the first use of a pleuroperitoneaI shunt in chylothorax in infants, with good results. We have inserted one in the left side of the chest and the peritoneal cavity. The technique of insertion has been described previously," The unique design of the shunt allows one-way flow of fluid from the pleural space to the peritoneal cavity, with low risk of flow failure, and compression of the pump chamber for the first 24 to 48 hours ensures its patency. The patient and family members are taught to pump about 100 times a day, which will move about 200 to 500 m1 of fluid a day in addition to the fluid that moves spontaneously. Median sternotomy and exploration of the pericardial space was considered but abandoned because of probable anatomical inaccessibility to the leaking lymphatic duct by this approach. In conclusion, our experience suggests that pleuroperitoneal shunting, which provides good palliation with minimal morbidity, may be the treatment of choice for this intractable chylothorax and possibly should have been used before transthoracic ligation of the thoracic duct. I.
2. 3.
4.
REFERENCES Thomas CS Jr, McGoon DC. Isolated massive chylopericardium following cardiopulmonary bypass. J THORAC CARDIOVASC SURG 1971 ;61 :945-8. Pollard WM, Schuchmann GF, Bowen TE. Isolated chylopericardium after cardiac operations. J THORAC CARDIOVASC SURG 1981 ;81 :943-6. Feteih W, Rao PS, Whisennand HH, Mardini MK, Lawrie GM. Chylopericardium: new complication of Blalock-Taussig-anastomosis, J THORAC CARDIOVASC SURG 1983;85:791-4. Schiessler A, John A, Pallua N, Bucherl ES. Chylopericardium following aorta-coronary bypassprocedure. Thorae Cardiovasc Surg 1984;32:112-4.
5. Bakay C, Wijers TS. Treatment of cardiac tamponade due to isolated chylopericardium following open heart surgery. J Cardiovasc Surg 1984;25:249-51. 6. Azizkhan RG, Canfield J, Alford BA, Rodgers BM. Pleuroperitoneal shunts in management of neonatal chylothorax. J Pediatric Surg 1983;18:842-50. 7. Milsom JW, Kron IL, Rheuben KS, Rodgers 8M. Chylothorax: an assessment of current surgical management. J THORAC CARDIOVASC SURG 1985;89:221-7. 8. WeeseJL, Schouten JT. Pleural peritoneal shunts for the treatment of malignant pleural effusion. Surg Gynecol Obstet 1982;154:391-2.
Malignant fibrous histiocytoma of left atrium Jan Lee, M.B., F.R.C.S., K. L. Cheung, M.B., M.R.C.P., Rebecca Wang, M.B., M.R.C.P., C. K. Mok, M.B., F.R.C.S., and M. Aung Khin, M.B., Ph.D., Hong Kong From the Cardiothoracic Division, University Surgical Unit, Hong Kong University, Grantham Hospital, Hong Kong. A case is presented of malignant fibrous histiocytoma arising adjacent to a mitral Carpentier-Edwards bioprosthesis placed 6 years previously. Sarcoma has been known to develop around implanted prosthetic foreign bodies. In the case reported, malignant fibrous histiocytoma developed at the site of a Carpentier-Edwards mitral bioprosthesis. Case report. A Chinese man, who had a closed mitral commissurotomy in 1972at the age of 33 years, wasadmitted with mitral restenosis in 1978. In October 1978 the mitral Address for reprints: Jan Lee, M.D., The Grantham Hospital, 125 Wong Chuk Hang Rd., Hong Kong.
Volume 94 Number 3 September 1987
valve was replaced with a Carpentier-Edwards bioprosthesis, * inserted with interrupted Teflon felt-supported 2-0 Ethibond sutures. At neither operation was anything unusual noted about the native mitral valve. However, during the second operation, thrombus was found in the left atrium. Histologic studies of the resected mitral valve showed chronic rheumatic valvulitis with calcification, and the thrombotic material was organizing thrombus. Warfarin was given for 1 year and then discontinued. The patient remained well until November 1984, when he had a transient episode of left hemiparesis for which warfarin was restarted. He rapidly became symptomatic over the next few weeks, and mitral regurgitation was detected. Despite the absence of fever and repeated negative blood cultures, he was treated for infective endocarditis because of the clinical picture and a grossly elevated white count of 70,OOO/mm J • An emergency operation was performed on Dec. 22, 1984, for uncontrollable pulmonary edema and a deteriorating level of consciousness. At operation, the left atrium was filled with clots. The Carpentier-Edwards bioprosthesis was still well preserved. Vegetation-like material was present at the anterior commissural area, the erosion of which had caused a severe periprosthetic leak. The bioprosthesis was replaced with a Medtronic Hall mitral prostheis. The man died 80 days later, never having regained consciousness. Pathologic findings. Necropsy revealed a markedly enlarged heart. The left atrium was 10 em in its largest diameter and was filled with clots. There were irregular grayish masses around the prosthetic mitral valve and on the atrial wall, infiltrating its full thickness and eroding into the pericardium and surrounding structures (Fig. 1). The brain was edematous with areas of softening and infiltration by grayish masses. No obvious primary tumor was found in other parts of the body including the extremities, skin, chest wall, and retroperitoneum. Microscopic examination of the masses (Fig. 2) revealed a cellular tumor consisting of sheets of closely packed pleomorphic spindle cells with oval vesicular nuclei containing one or more nucleoli, which formed fascicles and storiform patterns. Round or polygonal mononuclear cells with abundant foamy cytoplasm resembling histiocytes were present as were a few scattered multinucleated giant cells. Numerous mitotic figures were seen. The brain masses showed similar features consistent with malignant fibrous histiocytoma. Electron microscopic examination supported the diagnosis, showing mostly fibroblastic cells, histiocytoid cells, and undifferentiated mesenchymal cells.
Discussion Malignant fibrous histiocytoma occurs most commonly in the extremities, abdominal cavity, and retroperitoneum.' Thoracic malignant fibrous histiocytoma involving the lungs, mediastinum, and pericardium, although rare, has also been described." Malignant fibrous histiocytoma arising from the heart is very rare. So far, fewer than 20 cases had been reported.v'? Recently, Holtzman and colleagues" "Thesewing ringof the bioprosthesis had a softsilicone Rubberinsert covered witha porous, seamless polytetrafluoroethylene cloth. The chemical solution used in the fixation process was 0.625% glutaraldehyde in phosphate-buffered saline, pH of 7.4.The valve was finally packaged in fresh glutaraldehyde. (From American Edwards Laboratories, Santa Ana, Calif.)
Brief communications 4 5 I
Fig. 1. Left atrium showing irregular tumor masses around prosthetic mitral valve and on surrounding atrial wall.
Fig. 2. Microscopic appearance of atrial tumor showing storiform pattern of spindle and foamy cells. (Original magnification XlOO.)
reported a case of malignant fibrous histiocytoma in which the clinical course was very similar to that of our patient. Their patient had congestive heart failure, weakness, weight loss, and multiple embolic events 8 years after mitral valve replacement with a StarrEdwards M3 valve. The patient died 1 year later. Autopsy revealed a polypoid tumor adjacent to the anulus fibrosus of the prosthetic valve. Histologic features of the tumor were those of malignant fibrous histiocytoma. No other sites of tumor were found. Our case thus constitutes the second case of malignant fibrous histiocytoma complicating mitral valve replacement. Experimental work and clinical material have already shown that sarcoma can develop from insertion of prosthetic foreign bodies in tissues." Sarcoma had been reported arising from a pacemaker pocket, adjacent to Dacron vascular grafts, and around a metal implant for femur fracture. 13-15 If we implicate an
The Journal of
452
Brief communications
etiologic relationship between malignant fibrous histiocytoma and prosthetic valves, the carcinogenicity of the latter must be extremely low, because only two cases of this disease have been discovered from among hundreds of thousands of valve replacements over a quarter of a century. Only diligent reporting of such rare complications after valve replacement will shed more light on this interesting but rare condition. We thank Dr. I. T. M. Kung, Pathology Department, Hong Kong University, for electron microscopic studies of the tumor.
Thoracic and Cardiovascular Surgery
associated with a vascular prosthesis: a case report. Cancer 1980;46:398-402. 15. Lee YS, Pho RWH, Nather A. Malignant fibrous histiocytoma at the site of metal implant. Cancer 1984; 53:2286-9.
Mucoepidermoid bronchial adenoma in a 6-year-old girl: A case report and review of the literature Robert L. Archer, D.O., Stanley E. Grogg, D.O., and Steve P. Sanders, D.O., Tulsa. Okla.
REFERENCES I. Weiss S, Enzinger FM. Malignant fibrous histiocytoma:
2.
3.
4.
5.
6.
7.
8. 9.
10.
11.
12. 13. 14.
an analysis of 200 cases. Cancer 1978;41 :2250-66. Kern WH, Hughes RK, Meyer BW, Harley DP. Malignant fibrous histiocytoma of the lung. Cancer 1979; 44:1793-1801. Mills SA, Breyer RH, Johnston FR, et al. Malignant fibrous histiocytoma of the mediastinum and lung. J THORAC CARDIOVASC SURG 1982;84:367-72. Venn GE, Gellister J, DaCosta PE, Goldstraw P. Malignant fibrous histiocytoma in thoracic surgical practice. J THORAC CARDIOVASC SURG 1986;91:234-7. Mittal KR, Chopra P. Primary fibrous histiocytoma of the pericardium. Indian J Pathol Microbiol 1982;25:15760. Shah AA, Churg A, Sbarbaro JA, Sheppard JM, Lamberti J. Malignant fibrous histiocytoma of the heart presenting as an atrial myxoma. Cancer 1978; 42:246671. Terashima K, Aoyama K, Nihei K, et al. Malignant fibrous histiocytoma of the heart. Cancer 1983; 52:191926. Concha M, Pasalodos J, Rubio FL. Surgical treatment of primary malignant fibrous histiocytoma of the left atrium. Int J Cardiol 1983;4:463-6. Eckstein R, Gossner W, Rienmuller R. Primary malignant fibrous histiocytoma of the left atrium: surgical and chemotherapeutic management. Br Heart J 1984;52: 354-7. Kern SE, Cowen ME, Abrams GD. Malignant fibrous histiocytoma of the heart presenting as unilateral pulmonary thromboembolism and infarct. Hum Pathol 1985; 16:1279-81. Holtzman E, Schiby G, Segal P, Priel I. Malignant fibrous histiocytoma complicating mitral valve replacement. J Am Coll Cardiol 1986;7:956-60. Brand KG. Foreign body induced sarcoma. Becker FF, ed. Cancer, vol I. New York: Plenum Press, 1975:485. Fraedrich G, Kracht J, Scheid HH. Sarcoma of the lung in a pacemaker pocket-simple coincidence or oncotaxis. Thorac Cardiovasc Surg 1984;32:67. Weinberg DS, Maini BS. Primary sarcoma of the aorta
From the Departments of Cardiovascular Thoracic Surgery and Pediatrics, Oklahoma Osteopathic Hospital, Tulsa, Okla.
A rare case of a mucoepidermoid bronchialadenomadiscovered in a 6-year-old girl with recurrent pneumonia is presented. A review of the literature suggests that less than 15 cases in the pediatric age group have been reported. Although primary lung carcinomas are rare in children. this case illustrates that recurrent lobar pneumonia in children should be aggressively investigated and that the proper therapy is conservative surgical resection.
The rarity of all primary lung tumors in children has been substantiated in the literature; in children under the age of 14 only 15 cases were documented up to 1951. 1 Current references indicate that the incidence has not substantially increased.' Bronchial adenomas, which have been traditionally subcategorized as carcinoid, adenoid cystic (cylindroma), and mucoepidermoid, form the majority of bronchial neoplasms in children. Most series show that the mucoepidermoid form constitutes only 2.5%3 of these adenomas and that they have low-grade malignant potential." Until 1973 only 55 cases of mucoepidermoid tumors of the lung had been reported in any age group. 7,8 In children under the age of 14 years, only 12 cases of this particular tumor had been reported as of 1984. 9 An additional finding and surgical approach to therapy is discussed. Case report. A 6-year-old white girl was admitted for evaluation and treatment of pneumonia. There was a historyof pneumonia occurring 3 weeks before this admission, which had been treated with oral antibiotics and apparently had resolved. Two days before this admission a fever and cough developed. Radiographs revealedcomplete consolidation of the right middle lobe and partial consolidation of the right lower Address for reprints: Dr. S. P. Sanders, 5501 E. 48th Place, Tulsa, Oklo.. 74135.
4 5 0 Brief communications
Table I. Chylopericardium after CABG operation Patient (age. sex)
Operation
Cause
Pollard et al.' (1981)
57, M
CABGx4
Schiessler et al." (1984) Bakayet al.' (1984)
45, M 41, M
CABGX I CABGX3
Yes Yes
Lee et al. (1987)
49, M
CABGX
No
Left subclavian line Not known Left jugular line Not known
Authors
I
Treatment
Drainage, diet Drainage, diet Subxiphoid pericardioperitoneal fistula Drainage, diet, ligation thoracic duct, pleuroperitoneal shunt
Legend: CABG, Coronary artery bypass graft. CPB, Cardiopulmonary bypass.
fluid accumulation. The follow-up chest film taken 1 year later showed no abnormalities.
Discussion. Azizkhan," Milsom,' and their associates reported the first use of a pleuroperitoneaI shunt in chylothorax in infants, with good results. We have inserted one in the left side of the chest and the peritoneal cavity. The technique of insertion has been described previously," The unique design of the shunt allows one-way flow of fluid from the pleural space to the peritoneal cavity, with low risk of flow failure, and compression of the pump chamber for the first 24 to 48 hours ensures its patency. The patient and family members are taught to pump about 100 times a day, which will move about 200 to 500 m1 of fluid a day in addition to the fluid that moves spontaneously. Median sternotomy and exploration of the pericardial space was considered but abandoned because of probable anatomical inaccessibility to the leaking lymphatic duct by this approach. In conclusion, our experience suggests that pleuroperitoneal shunting, which provides good palliation with minimal morbidity, may be the treatment of choice for this intractable chylothorax and possibly should have been used before transthoracic ligation of the thoracic duct. I.
2. 3.
4.
REFERENCES Thomas CS Jr, McGoon DC. Isolated massive chylopericardium following cardiopulmonary bypass. J THORAC CARDIOVASC SURG 1971 ;61 :945-8. Pollard WM, Schuchmann GF, Bowen TE. Isolated chylopericardium after cardiac operations. J THORAC CARDIOVASC SURG 1981 ;81 :943-6. Feteih W, Rao PS, Whisennand HH, Mardini MK, Lawrie GM. Chylopericardium: new complication of Blalock-Taussig-anastomosis, J THORAC CARDIOVASC SURG 1983;85:791-4. Schiessler A, John A, Pallua N, Bucherl ES. Chylopericardium following aorta-coronary bypassprocedure. Thorae Cardiovasc Surg 1984;32:112-4.
5. Bakay C, Wijers TS. Treatment of cardiac tamponade due to isolated chylopericardium following open heart surgery. J Cardiovasc Surg 1984;25:249-51. 6. Azizkhan RG, Canfield J, Alford BA, Rodgers BM. Pleuroperitoneal shunts in management of neonatal chylothorax. J Pediatric Surg 1983;18:842-50. 7. Milsom JW, Kron IL, Rheuben KS, Rodgers 8M. Chylothorax: an assessment of current surgical management. J THORAC CARDIOVASC SURG 1985;89:221-7. 8. WeeseJL, Schouten JT. Pleural peritoneal shunts for the treatment of malignant pleural effusion. Surg Gynecol Obstet 1982;154:391-2.
Malignant fibrous histiocytoma of left atrium Jan Lee, M.B., F.R.C.S., K. L. Cheung, M.B., M.R.C.P., Rebecca Wang, M.B., M.R.C.P., C. K. Mok, M.B., F.R.C.S., and M. Aung Khin, M.B., Ph.D., Hong Kong From the Cardiothoracic Division, University Surgical Unit, Hong Kong University, Grantham Hospital, Hong Kong. A case is presented of malignant fibrous histiocytoma arising adjacent to a mitral Carpentier-Edwards bioprosthesis placed 6 years previously. Sarcoma has been known to develop around implanted prosthetic foreign bodies. In the case reported, malignant fibrous histiocytoma developed at the site of a Carpentier-Edwards mitral bioprosthesis. Case report. A Chinese man, who had a closed mitral commissurotomy in 1972at the age of 33 years, wasadmitted with mitral restenosis in 1978. In October 1978 the mitral Address for reprints: Jan Lee, M.D., The Grantham Hospital, 125 Wong Chuk Hang Rd., Hong Kong.
Volume 94 Number 3 September 1987
valve was replaced with a Carpentier-Edwards bioprosthesis, * inserted with interrupted Teflon felt-supported 2-0 Ethibond sutures. At neither operation was anything unusual noted about the native mitral valve. However, during the second operation, thrombus was found in the left atrium. Histologic studies of the resected mitral valve showed chronic rheumatic valvulitis with calcification, and the thrombotic material was organizing thrombus. Warfarin was given for 1 year and then discontinued. The patient remained well until November 1984, when he had a transient episode of left hemiparesis for which warfarin was restarted. He rapidly became symptomatic over the next few weeks, and mitral regurgitation was detected. Despite the absence of fever and repeated negative blood cultures, he was treated for infective endocarditis because of the clinical picture and a grossly elevated white count of 70,OOO/mm J • An emergency operation was performed on Dec. 22, 1984, for uncontrollable pulmonary edema and a deteriorating level of consciousness. At operation, the left atrium was filled with clots. The Carpentier-Edwards bioprosthesis was still well preserved. Vegetation-like material was present at the anterior commissural area, the erosion of which had caused a severe periprosthetic leak. The bioprosthesis was replaced with a Medtronic Hall mitral prostheis. The man died 80 days later, never having regained consciousness. Pathologic findings. Necropsy revealed a markedly enlarged heart. The left atrium was 10 em in its largest diameter and was filled with clots. There were irregular grayish masses around the prosthetic mitral valve and on the atrial wall, infiltrating its full thickness and eroding into the pericardium and surrounding structures (Fig. 1). The brain was edematous with areas of softening and infiltration by grayish masses. No obvious primary tumor was found in other parts of the body including the extremities, skin, chest wall, and retroperitoneum. Microscopic examination of the masses (Fig. 2) revealed a cellular tumor consisting of sheets of closely packed pleomorphic spindle cells with oval vesicular nuclei containing one or more nucleoli, which formed fascicles and storiform patterns. Round or polygonal mononuclear cells with abundant foamy cytoplasm resembling histiocytes were present as were a few scattered multinucleated giant cells. Numerous mitotic figures were seen. The brain masses showed similar features consistent with malignant fibrous histiocytoma. Electron microscopic examination supported the diagnosis, showing mostly fibroblastic cells, histiocytoid cells, and undifferentiated mesenchymal cells.
Discussion Malignant fibrous histiocytoma occurs most commonly in the extremities, abdominal cavity, and retroperitoneum.' Thoracic malignant fibrous histiocytoma involving the lungs, mediastinum, and pericardium, although rare, has also been described." Malignant fibrous histiocytoma arising from the heart is very rare. So far, fewer than 20 cases had been reported.v'? Recently, Holtzman and colleagues" "Thesewing ringof the bioprosthesis had a softsilicone Rubberinsert covered witha porous, seamless polytetrafluoroethylene cloth. The chemical solution used in the fixation process was 0.625% glutaraldehyde in phosphate-buffered saline, pH of 7.4.The valve was finally packaged in fresh glutaraldehyde. (From American Edwards Laboratories, Santa Ana, Calif.)
Brief communications 4 5 I
Fig. 1. Left atrium showing irregular tumor masses around prosthetic mitral valve and on surrounding atrial wall.
Fig. 2. Microscopic appearance of atrial tumor showing storiform pattern of spindle and foamy cells. (Original magnification XlOO.)
reported a case of malignant fibrous histiocytoma in which the clinical course was very similar to that of our patient. Their patient had congestive heart failure, weakness, weight loss, and multiple embolic events 8 years after mitral valve replacement with a StarrEdwards M3 valve. The patient died 1 year later. Autopsy revealed a polypoid tumor adjacent to the anulus fibrosus of the prosthetic valve. Histologic features of the tumor were those of malignant fibrous histiocytoma. No other sites of tumor were found. Our case thus constitutes the second case of malignant fibrous histiocytoma complicating mitral valve replacement. Experimental work and clinical material have already shown that sarcoma can develop from insertion of prosthetic foreign bodies in tissues." Sarcoma had been reported arising from a pacemaker pocket, adjacent to Dacron vascular grafts, and around a metal implant for femur fracture. 13-15 If we implicate an
The Journal of
452
Brief communications
etiologic relationship between malignant fibrous histiocytoma and prosthetic valves, the carcinogenicity of the latter must be extremely low, because only two cases of this disease have been discovered from among hundreds of thousands of valve replacements over a quarter of a century. Only diligent reporting of such rare complications after valve replacement will shed more light on this interesting but rare condition. We thank Dr. I. T. M. Kung, Pathology Department, Hong Kong University, for electron microscopic studies of the tumor.
Thoracic and Cardiovascular Surgery
associated with a vascular prosthesis: a case report. Cancer 1980;46:398-402. 15. Lee YS, Pho RWH, Nather A. Malignant fibrous histiocytoma at the site of metal implant. Cancer 1984; 53:2286-9.
Mucoepidermoid bronchial adenoma in a 6-year-old girl: A case report and review of the literature Robert L. Archer, D.O., Stanley E. Grogg, D.O., and Steve P. Sanders, D.O., Tulsa. Okla.
REFERENCES I. Weiss S, Enzinger FM. Malignant fibrous histiocytoma:
2.
3.
4.
5.
6.
7.
8. 9.
10.
11.
12. 13. 14.
an analysis of 200 cases. Cancer 1978;41 :2250-66. Kern WH, Hughes RK, Meyer BW, Harley DP. Malignant fibrous histiocytoma of the lung. Cancer 1979; 44:1793-1801. Mills SA, Breyer RH, Johnston FR, et al. Malignant fibrous histiocytoma of the mediastinum and lung. J THORAC CARDIOVASC SURG 1982;84:367-72. Venn GE, Gellister J, DaCosta PE, Goldstraw P. Malignant fibrous histiocytoma in thoracic surgical practice. J THORAC CARDIOVASC SURG 1986;91:234-7. Mittal KR, Chopra P. Primary fibrous histiocytoma of the pericardium. Indian J Pathol Microbiol 1982;25:15760. Shah AA, Churg A, Sbarbaro JA, Sheppard JM, Lamberti J. Malignant fibrous histiocytoma of the heart presenting as an atrial myxoma. Cancer 1978; 42:246671. Terashima K, Aoyama K, Nihei K, et al. Malignant fibrous histiocytoma of the heart. Cancer 1983; 52:191926. Concha M, Pasalodos J, Rubio FL. Surgical treatment of primary malignant fibrous histiocytoma of the left atrium. Int J Cardiol 1983;4:463-6. Eckstein R, Gossner W, Rienmuller R. Primary malignant fibrous histiocytoma of the left atrium: surgical and chemotherapeutic management. Br Heart J 1984;52: 354-7. Kern SE, Cowen ME, Abrams GD. Malignant fibrous histiocytoma of the heart presenting as unilateral pulmonary thromboembolism and infarct. Hum Pathol 1985; 16:1279-81. Holtzman E, Schiby G, Segal P, Priel I. Malignant fibrous histiocytoma complicating mitral valve replacement. J Am Coll Cardiol 1986;7:956-60. Brand KG. Foreign body induced sarcoma. Becker FF, ed. Cancer, vol I. New York: Plenum Press, 1975:485. Fraedrich G, Kracht J, Scheid HH. Sarcoma of the lung in a pacemaker pocket-simple coincidence or oncotaxis. Thorac Cardiovasc Surg 1984;32:67. Weinberg DS, Maini BS. Primary sarcoma of the aorta
From the Departments of Cardiovascular Thoracic Surgery and Pediatrics, Oklahoma Osteopathic Hospital, Tulsa, Okla.
A rare case of a mucoepidermoid bronchialadenomadiscovered in a 6-year-old girl with recurrent pneumonia is presented. A review of the literature suggests that less than 15 cases in the pediatric age group have been reported. Although primary lung carcinomas are rare in children. this case illustrates that recurrent lobar pneumonia in children should be aggressively investigated and that the proper therapy is conservative surgical resection.
The rarity of all primary lung tumors in children has been substantiated in the literature; in children under the age of 14 only 15 cases were documented up to 1951. 1 Current references indicate that the incidence has not substantially increased.' Bronchial adenomas, which have been traditionally subcategorized as carcinoid, adenoid cystic (cylindroma), and mucoepidermoid, form the majority of bronchial neoplasms in children. Most series show that the mucoepidermoid form constitutes only 2.5%3 of these adenomas and that they have low-grade malignant potential." Until 1973 only 55 cases of mucoepidermoid tumors of the lung had been reported in any age group. 7,8 In children under the age of 14 years, only 12 cases of this particular tumor had been reported as of 1984. 9 An additional finding and surgical approach to therapy is discussed. Case report. A 6-year-old white girl was admitted for evaluation and treatment of pneumonia. There was a historyof pneumonia occurring 3 weeks before this admission, which had been treated with oral antibiotics and apparently had resolved. Two days before this admission a fever and cough developed. Radiographs revealedcomplete consolidation of the right middle lobe and partial consolidation of the right lower Address for reprints: Dr. S. P. Sanders, 5501 E. 48th Place, Tulsa, Oklo.. 74135.