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Multiple Bronchial Polyps
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MULTIPLE BRONCHIAL POLYPS bilaterally. The remainder of the postoperative course was uneventful, and following transfer to the Institute of Rehahilitation Medicine at New York University, the patient is able to hrace walk with assistance. DISCUSSION
Most thoracic aortic aneurysms have a characteristic appearance on chest x-ray film. Those of the descending aorta usually project to the left and posteriorly. The projection of such an aneurysm to the right of midline is unusual, and it was not until 1949 that the first case report" appeared. Subsequently there have been several isolated case reportsv" which have illustrated how easily a mistaken diagnosis of bronchogenic carcinoma can be made. The present case demonstrates the advantage of tomography in delineating the outline of right sided thoracic aortic aneurysms (Fig 2) and thus differentiating them from other paramediastinal masses. The diagnosis of aneurysm can most readily be made by the use of aortography, whether by employing retrograde catheterization or by observing the delayed phase of a venous angiocardiogram. The operative technique used in this easel is a satisfactory method for resection of a saccular aneurysm of the descending thoracic aorta. The importance of a simple approach and the elimination of the need for anticoagulation cannot be underestimated. Distal aortic perfusion was well maintained, as determined by adequacy of urine output, despite a drop of 50 mm Hg pressure across the shunt from proximal to distal aorta. The development of a spinal cord ischemic lesion is unusual following only severance of intercostal arteries without aortic occlusion as in the present case. Adams and van Ceertruyden" analyzed the neurologic complications after aortic surgery and were able to find only one similar case report. They attribute this spinal ischemia to the presence of a high arteria radicularis magna which is sacrificed during aortic surgery, resulting in a lesion of the lower one-fourth of the spinal cord. The arteria radicularis magna is the lowest and best developed radicularis artery and may arise from the eighth thoracic to the fourth lumbar intercostal artery. Even in the event that the radicularis magna is sacrificed, spinal damage may not necessarily occur because of the presence of a collateral blood supply which would probably have been adequate in a less atherosclerotic patient than ours. In addition, the effect of significant drug-induced hypotension during the immediate postoperative period is difficult to evaluate, but most likely it also played an important part in the development of spinal ischemia. CHEST, VOL. 57, NO.3, MARCH 1970
REFERENCES
2
3 4 5
6
KAHN, D.R., VATHAYANON, S. AND SLOAN, H.: Resection of descending thoracic aneurysms without left heart bypass, Arch. Surg., 97:336, 1968. EpSTEIN, B.S. AND FRIEDMAN, R.L.: Arteriosclerotic aneurysm of the descending thoracic aorta presenting to the right of the spine, Radiology, 53:93, 1949. DAVIS, J.G. AND WINSOR, P.: Roentgen findings in aneurysm of the descending thoracic aorta, Amer J. Roentgenol., 81:819, 1959. MALCOLM, J.A., LAWRENCE, L.R. AND PRINCIPATO, D.J.: Aortic aneurysms of right side, New York State J. Med., 67:1772,1967. CLAXTON, C.P. AND DILLON, JR., M.L.: Saccular aneurysm of the descending thoracic aorta presenting as a right chest mass, Ann. Thorac. Surg., 7:34, 1969. ADAMS, H.D., AND VAN GEERTRUYDEN, H.H.: Neurologic complications of aortic surgery, Ann. Surg., 144:574, 1956.
Reprint requests: Dr. Engelman, New York University School of Medicine, 550 First Avenue, New York, New York 10016.
Multiple Bronchial Polyps Bertram Kahn, M.D., and Norman S. Amer, M.D., F.G.G.P.
A patient thought to be suffering from bronchial asthma for almost ten years finally required hospitalization because of signs suggestive of bronchogenic carcinoma. Surprisingly, multiple benign polyps were found in the left lower lobe bronchus, and bronchoscopic extirpation cured the "carcinoma" and the "asthma" as well.
Multiple bronchial polyps are a rare finding, but in spite of their infrequent occurrence, they must be considered in the differential diagnosis of partial or complete obstruction of the tracheobronchial tree. Whereas bronchogenic carcinoma, the most common neoplasm associated with such obstruction, is highly malignant, bronchial polyps are benign. Thus, the therapeutic management of each is considerably different. The following case report shows that a benign bronchial tumor can be eradicated completely by means of bronchoscopic extirpation without the need for thoracotomy. Further, it is demonstrated that a complete differential diagnosis must be exhausted in all patients who present with bronchial obstruction, for if only this be done, then individuals can be saved from unnecessary major surgery with its consequent high risk of morbidity and mortality. CASE REPORT
A 65-year-old white woman was admitted to the Peninsula General Hospital, Queens, New York, on January 7, 1966 with a diagnosis of left lobe pneumonia. Her main symptoms
280 had been present for three weeks prior to admission and consisted of chills, fever, slight malaise, dyspnea, wheezing and cough with expectoration of yellow sputum. Shortly after the beginning of her illness, a chest x-ray examination demonstrated left lower lobe infiltrate. Antimicrobial agents, bronchodilators and expectorants were administered, but symptoms persisted. Four days prior to admission, a followup chest x-ray film showed progression of the infiltrate with atelectasis of the left lower lobe. The mediastinum was shifted to the left and the left diaphragm was elevated. Since the findings suggested bronchial obstruction, possibly due to carcinoma, hospitalization was felt necessary for diagnostic evaluation and appropriate therapy. The past history was interesting in that the patient had been treated for bronchial asthma since she was 55 years old. Wheezing had never heen experienced during her younger years, there being no history of childhood asthma and no history of familial or personal atopy. The patient did undergo a complete allergy skin test study, but there was no positive reaction of significance. The diagnosis of asthma was assumed because of the presence of "lots of wheezing," "recurrent chronic bronchitis" and the apparent beneficial response to nebulized and oral bronchodilators. There was no history of cardiac disease and cardiac medications had never been prescribed. There was also no history of exposure to tuberculosis, pulmonary fungal infections, or to various dusts, fumes or industrial irritants, and no history of smoking. There was also no exposure to household pets, and environmental changes were limited as the patient never traveled outside the New York state area. Aside from the pulmonary problem, the patient also suffered from Parkinson's disease, but this was of a mild nature and fairly well controlled by drug treatment. For a period of four months prior to hospitalization, there had been a considerable loss of appetite and a 30 pound weight loss. There were no gastrointestinal, genitourinary, or musculoskeletal
Fn;uHE 1. Chest roentgenogram on admission to hospital showing opacification and loss of aeration of the left lower lung field with elevation of the diapragm and mediastinal shift to the left.
KAHN AND AMER
FIGURE 2. Three distinct and separate polyps extirpated through the bronchoscope from the left lower lobe bronchus on January 25, 1966.
complaints of recent origin, and the patient felt well in all other respects. Physical examination revealed an alert, intelligent, welloriented woman in moderate respiratory distress. An apparent tremor on motion of the extremities was noted and this was felt to be consistent with Parkinson's disease. On admission, she was febrile, having a temperature of 101 0 F. Nasal examination failed to show any evidence of polyps. There was no evidence of lymphadenopathy in the neck, but the trachea was shifted slightly to the left. Chest inspection revealed the narrow interspaces and contraction of the rib cage on the left side. Percussion sounds over the left lower lung field were dull, and breath sounds were absent posteriorly in the same area. Sibilant rales were audible throughout the chest except over the left lower lung field, and wheezing appeared to be louder over the left upper chest. The admission chest x-ray film (Fig 1) showed elevation of the left diaphragm, narrowing of the left intercostal spaces, mediastinal shift to the left, and opacification with loss of aeration of the left lower lung field. The initial impression was that the patient had atelectasis due to carcinomatous bronchial obstruction. The previous management with antimicrobial agents, bronchodilators and expectorants was continued. In addition, intermittent positive pressure breathing therapy was also started. The patient's response was excellent as the chills and fever resolved completely, and there was a return of appetite and state of well-being. However, atelectasis persisted. Laboratory studies included Papanicolaou sputum cytology, roentgen bone survey, and various blood determinations, but all of these gave normal results. Three attempts at thoracentesis of a suspected left sided pleural effusion failed to produce any fluid. Bronchography revealed a pale graypink friable tumor mass projecting on a broad base from the posterolateral wall of the left lower lobe bronchial lumen, and lying just distal to the orifice of the superior segment. This lesion was the apparent cause of the obstruction and secondary atelectasis since it filled almost 90 percent of the lumen on inspiration and 100 percent after expiration. Although the tumor was thought to be a polypoid bronchogenic carcinoma, histologic examination surprisingly showed a benign connective tissue neoplasm with fibrous stroma and areas of hyalinization and edema. Thus, the lesion was a benign fibroma or polyp, and not a bronchogenic carcinoma. In view of the pathologic findings, hronchoscopy was repeated the following week, in order that residual tumor
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MULTIPLE BRONCHIAL POLYPS could be excised. Following removal of the original 1.0 em x 1.0 cm stop-valve polypoid tumor (Fig 2), two additional polyps (Fig 2), 2.5 cm x 1.2 cm and 3.0 cm x 1.5 em, respectively, were observed and these were also extirpated through the bronchoscope. Each of these latter two polyps had been attached to the bronchial wall by a 0.5 cm pedicle. During final withdrawal of the examining scope, the distal basal segmental orifices could not be visualized because of the technical problem created by the friable raw bleeding surfaces of the multiple polyps. Microscopically (Fig 3) the polypoid masses were lined by slightly hyperplastic columnar epithelium, and their supporting stroma consisted of edematous loose areolar tissue. Although they bore a strong resemblance to nasal polyps, the bronchial lesions contained few eosinophils. Instead, the infiltrate consisted primarily of lymphocytes and plasma cells. Thus, the tumors were benign bronchial mucosal polyps. Since follow-up roentgenographic studies of the chest failed to show resolution of the atelectasis, bronchoscopy was repeated once more, and another large polypoid mass was found, measuring 1.6 em x 1.2 cm (Fig 4) and lying on a 0.6 cm pedicle base. After its removal, the orifices of the basal segments of the left lower lobe could be visualized in full, and follow-up roentgenographic studies including a percutaneous transcricoid bronchogram revealed complete filling of the major basal bronchi of the left lower lobe. Tubular bronchiectasis and residual areas of segmental atelectasis, however, were evident, but these findings were without clinical significance as the use of antimicrobial agents, intermittent positive pressure ventilation, expectorants and other supportive medical therapy allowed the
FIGURE 3. Microscopic (100 x) appearance of polypoid mass showing slight hyperplastic columnar epithelium and edematous loose areolar tissue with lymphocytic and plasma cell infiltrate.
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FIGURE 4. Fourth and final bronchial polyp, also bronchoscopically extirpated, on January 31, 1966. patient to recover completely and leave the hospital on February 9, 1966. During the past three years of follow-up, chest x-ray examinations have shown persistent opacification of the left lower lobe, reflecting either a chronic pleural reaction or possibly segmental atelectasis. In spite of this, antibiotics, branchodilators and expectorants have not been needed and there has been no recurrence of wheezing or "asthma." In fact since her discharge from the hospital, the patient has yet to experience pulmonary symptoms. DISCUSSION
The pathologic finding of polyps in the bronchial tree is most unusual. Patterson! in his review in 1930 could find only five such cases in the medical literature, but since then, there have been sufficient reports which indicate that bronchial polyps are more common than previously appreciated. Notwithstanding, the finding of multiple polyps in the distal tracheobronchial tree is indeed rare, most case reports describing a single or isolated polypoid lesion in the more proximal portions of the upper respiratory tract, ie, in the trachea and upper bronchi. Fibroepithelial polyps, unlike other polypoid bronchial tumors, do not exhibit the squamous epithelial over-growth of papillomas nor the bizarre and varied patterns of bronchial adenomas," Instead, they consist of a loose connective tissue core covered by normal bronchial respiratory epithelium, the core resembling the myxomatous regions of edematous nasal polyps." Unlike nasal polyps, however, fibroepithelial polyps are not usually multiple and are not necessarily found only in atopic individuals. In addition, they do not usually contain eosinophils. One notable exception, however, is the recent report of a patient with longterm bronchial asthma who did indeed have an eosinophil rather than a sparse plasma cell-lymphocyte infiltrated bronchial polyp."
282 Our patient was also thought to have bronchial asthma, and she, in fact, was treated for this condition for ten years prior to hospitalization, but none of her polyps contained eosinophils, and she never suffered from nasal allergy nor did she ever have nasal polyps. She also failed to have a noteworthy family history of atopy, and thus far, during the past three years since her discharge from the hospital, there has not been any recurrence of wheezing. Thus, the diagnosis of bronchial asthma appeared to be an error, and the bouts of wheezing were probably on the basis of recurrent transient episodes of polyp induced stop-valve type bronchial obstruction.v" The pathogenesis of bronchial polyps is still unresolved, and it is not certain whether the inflammation is a primary event or a secondary result of obstruction.t-" Thus bronchial polyps may be categorized as either inflammatory or neoplastic (inflammatory-like). In our patient, the bronchial polyps appear to be secondary to obstruction, since wheezing had been present for a number of years prior to the onset of bronchial inflammation, and endoscopic extirpation of the obstruction resulted in complete cure without the need for further use of antimicrobial agents. Although there was no clear-cut evidence here that the polyp was the end result of chronic bronchial inflammatory disease, the existence of such polypoid chronic bronchitis is well documented.t-v'? One such case was reported a few years ago and concerned a 34-year-old woman who had a history of chronic bronchitis since childhood.!" She developed bronchial obstruction due to heaped up polypoid granular tissue in the lumen of the left lower lobe bronchus. Although bronchoscopic removal of the offending tissue relieved her, she was not considered cured in view of continued bronchitis, thus anticipating future recurrence. In the same report, a second case was presented involving a 62-year-old laborer in whom there was a three to four week history of dyspnea, cough, yellowgreen sputum expectoration, and right-sided plural effusion. Bronchoscopy revealed a 2 ern granulomatous mass obstructing the right lower lobe bronchus. Endoscopic removal resulted in complete cure of the patient, and pathologic study revealed that the mass was actually a polyp clothed by columnar ciliated epithelium. The polyp's core consisted of edematous fibrous tissue which was vascular and which also contained acute inflammatory infiltrate. In spite of the pathologic findings reported in this man, the histology of our patient's bronchial polyps and of those reported by others failed to show acute inflammatory cellular or eosinophilic invasion.
KAHN AND AMER
Rather, sparse numbers of lymphocytes and plasma cells have predominated in almost all cases. Von Eicken, in 1907, was the first to use a bronchoscope for the diagnosis and removal of a benign bronchial neoplasm; and in 1910 Speiss was the first to report the bronchoscopic removal of a fibrous polypoid tumor. n- Ia Since then there have been numerous reports in the literature describing complete cure of bronchial tumors by means of bronchoscopic extirpation.1,4·11.14-111 It is true that the advent of improved surgical techniques has also resulted in the successful elimination of benign bronchial tumors by means of pulmonary resection and bronchotomy.Y-!? However, such improved techniques and successful results do not refute the statement of Jackson and Jackson,20 in 1938, that "almost all benign endobronchial tumors and tumor-like formations can be removed bronchoscopically and the prognosis as to life in such cases is good". The present case report reinforces the statement and shows that the use of endoscopic techniques still deserves serious consideration by the thoracic surgeon. A three year follow-up of our patient confirms the apparent success of bronchoscopic extirpation of the offending obstruction, there being a return of normal pulmonary drainage and a restoration of the patient to good pulmonary health. The clinical significance of the present case, therefore, is three-fold. First, it is obvious that fibroepithelial polyps of the bronchus can be mistaken clinically for carcinoma and lead to serious therapeutic errors. Second, although these lesions are benign both in growth and microscopic anatomic appearance, significant obstruction may result in impaired drainage of bronchial secretions producing secondary infection and a respiratory symptom complex. Third, the operative procedure required for the removal of these lesions is relatively simple with few hazards involved, and striking relief of the respiratory symptom complex can result. REFERENCES
1 2 3
4 5
PATTERSON, E.G.: Benign bronchial neoplasms, Arch. Otolarung., 12:739, 1930. ROWLANDS, D.T., JR.: Fibroepithelial polyps of the bronchus: A case report and review of the literature, Dis. Chest, 37:199, 1960. CALDAROLA, V.T., HARRISON, E.G., JR., CLAGETT, O.T., AND SCHMIDT, H.\V.: Benign tumors and tumor-like conditions of the trachea and bronchi, Ann. Otolaryng., 73:1042, 1964. PERONI, A.: Inflammatory tumors of the bronchi, Arch. Otolaryng., 19:1, 1934. REISNER, D.: A case of intrabronchial polypoid adenoma, with a review of the literature, Arch. Surgery, 16:1201, 1928.
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FATAL SUBDURAL HEMATOMA 6 SAINI, V.K. AND WAKE, P.L.: Inflammatory polyp of the bronchus, Ann. Thor. Surg., 5:141, 1968. 7 SALEK, J., PAZDERKA, S., AND ZAK, F.: Solitary bronchial polyps of inflammatory origin: A report of 2 cases treated by operation, J. Thor. Surg., 35:807, 1958. 8 CITRONI, G.A., AND DI GUGLIELMO, L.: Inflammatory polypi of the bronchi, Minerva Med., 46:686,1955. 9 JACKSON, C., AND JACKSON, C.L.: Benign tumors of the trachea and bronchi, JAMA, 99:1747,1932. 10 ASHLEY, D.J.B., DANINO, E.A., AND DAVIES, H.D.: Bronchial polyps, Thorax, 18:45, 1963. 11 MYERSON, M.C.: Benign neoplasms of the bronchus: Report of a case of fibrolipoma of the left main bronchus removed through the hronchoscope, Amer. J. M. Sci., 176:720, 1928. 12 VON EIKEN, c.. Bronchoscopische mitteilungen, Verhandl. d. Vereim Sud. Dent. Laryngol., 1907,410. 13 SPEISS, G.: Eine fall von hochgraduger dyspnoe infolge eines polypen in r. bronchus, Munchen Med. Wchmchr., 1910, 4, 2095. 14 JESBERG, S., Bronchoscopic studies of some conditions of the respiratory tract, Ann. Otolaryng., 35:509, 1926. 15 LEMANN, I.I., Bronchial polyp associated with mediastinal mass, Med. Clin. of N.A., 12:1329, 1929. 16 LINDGREN, A.G.H.: Benignant polypous bronchial tumors, Acta. Otolaryng., 27:183, 1939. 17 PELEG, J., AND PAUZNER, Y.: Benign tumors of the lung, Dis. Chest, 47:179,1965. 18 YANKAUER, S.: Benign tumor of bronchus, Laryngoscope, 39:549, 1929. 19 ZAMORA, A.M.: Two benign growths removed by bronchoscopy, J. Laryng. and Otol. 46:829,1931. 20 JACKSON, C., AND JACKSON, C.L.: Benign tumors and tumor-like conditions in the tracheobronchial tree, Amer. J. Surg., 42:275, 1938. Reprint requests: Dr. Kahn, 1128 Medical Drive, Tyler, Texas 75701.
Fatal Subdural Hematoma: An Unusual Complication of Pacemaker Failure* Alfred Heilbrunn, M.D. O O
C6nical pacemaker failures are usually manifested by return of complete heart block, recurrence of StokesAdams attacks or sudden death. In the case presented, the patient sustained cerebral trauma during a StokesAdams attack, but symptoms of his subdural hematoma did not become evident until five days after re-establishment of pacing. The posslbDity of intracranial trauma should be considered fonowing Stokes-Adams attacks especially if changes in cerebral status occur.
pacemaker systems are generally acI mplantable cepted as definitive treatment for patients with symptomatic complete heart block.':" However, the
°From the Department of Surgery, Veterans Administration Hospital, Kansas City, Missouri and the University of Kansas School of Medicine, Kansas City, Kansas. ooChief, Surgical Service and Associate Professor of Surgery.
CHEST, VOL. 57, NO.3, MARCH 1970
unpredictability of battery life remains a problem. Opinion regarding replacement has varied considerably.':" While Chardack! recommends elective replacement at 30 months, Zo1l6 recommends replacement only because of malfunction or evidence of battery exhaustion. The following patient is of interest since he developed a previously unreported complication of pacemaker failure after 43 months of continuous pacing. CASE REPORT
An 82-year-old white man was admitted to the Kansas City Veterans Administration Hospital in October, 1967 with syncopal episodes due to pacemaker failure. The patient had transient complete heart block in 1955. Following the onset of Stokes-Adams attacks in October, 1962, a pacemaker with myocardial electrodes was inserted at another hospital. Failure of this pacemaker occurred in February, 1964 and it was replaced at our hospital by an Electrodyne pacemaker with myocardial electrodes. The patient was seen at regular intervals with pacing at 72 per minute and asymptomatic. In October, 1969, syncopal episodes recurred and the patient was returned to the hospital the following day. The pulse rate was 28 per minute. Pacing was re-established using a temporary transvenous catheter electrode. Because of his advanced age (82), it was felt inadvisable to attempt another thoracotomy. A Medtronic implantable transvenous pacemaker was obtained for insertion. While discussing the proposed procedure with the patient he complained of pain in the hip which had been aggravated during insertion of the temporary catheter electrode. Therefore, following implantation of the permanent pacemaker system, x-ray films of the hips were obtained. but no evidence of bony injury was seen. For the first five days following the implantation of the new unit, the patient did well, pacing at 73 beats per minute without extrasystoles or arrhythmia. He was then noted to be increasingly lethargic, his fluid intake had become inadequate and his blood urea nitrogen had risen to 38 mg percent. However, in spite of correction of fluid and electrolyte problems, the patient's level of consciousness continued to deteriorate during the next 36 hours. No localizing signs were present. A spinal tap showed xanthochromic fluid and the possibility of subdural hematoma was then considered. Bilateral carotid arteriography was undertaken and demonstrated hilateral space-occupying lesions. Following neurosurgical consultation, hilateral frontoparietal trephinations with evacuation of subdural hematoma from both sides was accomplished. Postoperatively, the patient showed some improvement, but never regained consciousness. His condition gradually deteriorated, and in spite of intensive supportive management, he developed pneumonia and expired in January, 1968. Throughout the prolonged postoperative course, there was no difficulty with the cardiac pacemaker system. Autopsy examination showed resolving bilateral subdural hematomas. There were old infarcts in both occipital lohes of the cerebrum and acute confluent bronchopneumonia of the lungs. The transvenous pacemaker electrode was in good position in the apex of the right ventricle and throughout most of its course had become covered with the smooth thin layer of tissue. At the point where the electrode traversed the tricuspid valve was an area of fibrous thickening.
MULTIPLE BRONCHIAL POLYPS bilaterally. The remainder of the postoperative course was uneventful, and following transfer to the Institute of Rehahilitation Medicine at New York University, the patient is able to hrace walk with assistance. DISCUSSION
Most thoracic aortic aneurysms have a characteristic appearance on chest x-ray film. Those of the descending aorta usually project to the left and posteriorly. The projection of such an aneurysm to the right of midline is unusual, and it was not until 1949 that the first case report" appeared. Subsequently there have been several isolated case reportsv" which have illustrated how easily a mistaken diagnosis of bronchogenic carcinoma can be made. The present case demonstrates the advantage of tomography in delineating the outline of right sided thoracic aortic aneurysms (Fig 2) and thus differentiating them from other paramediastinal masses. The diagnosis of aneurysm can most readily be made by the use of aortography, whether by employing retrograde catheterization or by observing the delayed phase of a venous angiocardiogram. The operative technique used in this easel is a satisfactory method for resection of a saccular aneurysm of the descending thoracic aorta. The importance of a simple approach and the elimination of the need for anticoagulation cannot be underestimated. Distal aortic perfusion was well maintained, as determined by adequacy of urine output, despite a drop of 50 mm Hg pressure across the shunt from proximal to distal aorta. The development of a spinal cord ischemic lesion is unusual following only severance of intercostal arteries without aortic occlusion as in the present case. Adams and van Ceertruyden" analyzed the neurologic complications after aortic surgery and were able to find only one similar case report. They attribute this spinal ischemia to the presence of a high arteria radicularis magna which is sacrificed during aortic surgery, resulting in a lesion of the lower one-fourth of the spinal cord. The arteria radicularis magna is the lowest and best developed radicularis artery and may arise from the eighth thoracic to the fourth lumbar intercostal artery. Even in the event that the radicularis magna is sacrificed, spinal damage may not necessarily occur because of the presence of a collateral blood supply which would probably have been adequate in a less atherosclerotic patient than ours. In addition, the effect of significant drug-induced hypotension during the immediate postoperative period is difficult to evaluate, but most likely it also played an important part in the development of spinal ischemia. CHEST, VOL. 57, NO.3, MARCH 1970
REFERENCES
2
3 4 5
6
KAHN, D.R., VATHAYANON, S. AND SLOAN, H.: Resection of descending thoracic aneurysms without left heart bypass, Arch. Surg., 97:336, 1968. EpSTEIN, B.S. AND FRIEDMAN, R.L.: Arteriosclerotic aneurysm of the descending thoracic aorta presenting to the right of the spine, Radiology, 53:93, 1949. DAVIS, J.G. AND WINSOR, P.: Roentgen findings in aneurysm of the descending thoracic aorta, Amer J. Roentgenol., 81:819, 1959. MALCOLM, J.A., LAWRENCE, L.R. AND PRINCIPATO, D.J.: Aortic aneurysms of right side, New York State J. Med., 67:1772,1967. CLAXTON, C.P. AND DILLON, JR., M.L.: Saccular aneurysm of the descending thoracic aorta presenting as a right chest mass, Ann. Thorac. Surg., 7:34, 1969. ADAMS, H.D., AND VAN GEERTRUYDEN, H.H.: Neurologic complications of aortic surgery, Ann. Surg., 144:574, 1956.
Reprint requests: Dr. Engelman, New York University School of Medicine, 550 First Avenue, New York, New York 10016.
Multiple Bronchial Polyps Bertram Kahn, M.D., and Norman S. Amer, M.D., F.G.G.P.
A patient thought to be suffering from bronchial asthma for almost ten years finally required hospitalization because of signs suggestive of bronchogenic carcinoma. Surprisingly, multiple benign polyps were found in the left lower lobe bronchus, and bronchoscopic extirpation cured the "carcinoma" and the "asthma" as well.
Multiple bronchial polyps are a rare finding, but in spite of their infrequent occurrence, they must be considered in the differential diagnosis of partial or complete obstruction of the tracheobronchial tree. Whereas bronchogenic carcinoma, the most common neoplasm associated with such obstruction, is highly malignant, bronchial polyps are benign. Thus, the therapeutic management of each is considerably different. The following case report shows that a benign bronchial tumor can be eradicated completely by means of bronchoscopic extirpation without the need for thoracotomy. Further, it is demonstrated that a complete differential diagnosis must be exhausted in all patients who present with bronchial obstruction, for if only this be done, then individuals can be saved from unnecessary major surgery with its consequent high risk of morbidity and mortality. CASE REPORT
A 65-year-old white woman was admitted to the Peninsula General Hospital, Queens, New York, on January 7, 1966 with a diagnosis of left lobe pneumonia. Her main symptoms
280 had been present for three weeks prior to admission and consisted of chills, fever, slight malaise, dyspnea, wheezing and cough with expectoration of yellow sputum. Shortly after the beginning of her illness, a chest x-ray examination demonstrated left lower lobe infiltrate. Antimicrobial agents, bronchodilators and expectorants were administered, but symptoms persisted. Four days prior to admission, a followup chest x-ray film showed progression of the infiltrate with atelectasis of the left lower lobe. The mediastinum was shifted to the left and the left diaphragm was elevated. Since the findings suggested bronchial obstruction, possibly due to carcinoma, hospitalization was felt necessary for diagnostic evaluation and appropriate therapy. The past history was interesting in that the patient had been treated for bronchial asthma since she was 55 years old. Wheezing had never heen experienced during her younger years, there being no history of childhood asthma and no history of familial or personal atopy. The patient did undergo a complete allergy skin test study, but there was no positive reaction of significance. The diagnosis of asthma was assumed because of the presence of "lots of wheezing," "recurrent chronic bronchitis" and the apparent beneficial response to nebulized and oral bronchodilators. There was no history of cardiac disease and cardiac medications had never been prescribed. There was also no history of exposure to tuberculosis, pulmonary fungal infections, or to various dusts, fumes or industrial irritants, and no history of smoking. There was also no exposure to household pets, and environmental changes were limited as the patient never traveled outside the New York state area. Aside from the pulmonary problem, the patient also suffered from Parkinson's disease, but this was of a mild nature and fairly well controlled by drug treatment. For a period of four months prior to hospitalization, there had been a considerable loss of appetite and a 30 pound weight loss. There were no gastrointestinal, genitourinary, or musculoskeletal
Fn;uHE 1. Chest roentgenogram on admission to hospital showing opacification and loss of aeration of the left lower lung field with elevation of the diapragm and mediastinal shift to the left.
KAHN AND AMER
FIGURE 2. Three distinct and separate polyps extirpated through the bronchoscope from the left lower lobe bronchus on January 25, 1966.
complaints of recent origin, and the patient felt well in all other respects. Physical examination revealed an alert, intelligent, welloriented woman in moderate respiratory distress. An apparent tremor on motion of the extremities was noted and this was felt to be consistent with Parkinson's disease. On admission, she was febrile, having a temperature of 101 0 F. Nasal examination failed to show any evidence of polyps. There was no evidence of lymphadenopathy in the neck, but the trachea was shifted slightly to the left. Chest inspection revealed the narrow interspaces and contraction of the rib cage on the left side. Percussion sounds over the left lower lung field were dull, and breath sounds were absent posteriorly in the same area. Sibilant rales were audible throughout the chest except over the left lower lung field, and wheezing appeared to be louder over the left upper chest. The admission chest x-ray film (Fig 1) showed elevation of the left diaphragm, narrowing of the left intercostal spaces, mediastinal shift to the left, and opacification with loss of aeration of the left lower lung field. The initial impression was that the patient had atelectasis due to carcinomatous bronchial obstruction. The previous management with antimicrobial agents, bronchodilators and expectorants was continued. In addition, intermittent positive pressure breathing therapy was also started. The patient's response was excellent as the chills and fever resolved completely, and there was a return of appetite and state of well-being. However, atelectasis persisted. Laboratory studies included Papanicolaou sputum cytology, roentgen bone survey, and various blood determinations, but all of these gave normal results. Three attempts at thoracentesis of a suspected left sided pleural effusion failed to produce any fluid. Bronchography revealed a pale graypink friable tumor mass projecting on a broad base from the posterolateral wall of the left lower lobe bronchial lumen, and lying just distal to the orifice of the superior segment. This lesion was the apparent cause of the obstruction and secondary atelectasis since it filled almost 90 percent of the lumen on inspiration and 100 percent after expiration. Although the tumor was thought to be a polypoid bronchogenic carcinoma, histologic examination surprisingly showed a benign connective tissue neoplasm with fibrous stroma and areas of hyalinization and edema. Thus, the lesion was a benign fibroma or polyp, and not a bronchogenic carcinoma. In view of the pathologic findings, hronchoscopy was repeated the following week, in order that residual tumor
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MULTIPLE BRONCHIAL POLYPS could be excised. Following removal of the original 1.0 em x 1.0 cm stop-valve polypoid tumor (Fig 2), two additional polyps (Fig 2), 2.5 cm x 1.2 cm and 3.0 cm x 1.5 em, respectively, were observed and these were also extirpated through the bronchoscope. Each of these latter two polyps had been attached to the bronchial wall by a 0.5 cm pedicle. During final withdrawal of the examining scope, the distal basal segmental orifices could not be visualized because of the technical problem created by the friable raw bleeding surfaces of the multiple polyps. Microscopically (Fig 3) the polypoid masses were lined by slightly hyperplastic columnar epithelium, and their supporting stroma consisted of edematous loose areolar tissue. Although they bore a strong resemblance to nasal polyps, the bronchial lesions contained few eosinophils. Instead, the infiltrate consisted primarily of lymphocytes and plasma cells. Thus, the tumors were benign bronchial mucosal polyps. Since follow-up roentgenographic studies of the chest failed to show resolution of the atelectasis, bronchoscopy was repeated once more, and another large polypoid mass was found, measuring 1.6 em x 1.2 cm (Fig 4) and lying on a 0.6 cm pedicle base. After its removal, the orifices of the basal segments of the left lower lobe could be visualized in full, and follow-up roentgenographic studies including a percutaneous transcricoid bronchogram revealed complete filling of the major basal bronchi of the left lower lobe. Tubular bronchiectasis and residual areas of segmental atelectasis, however, were evident, but these findings were without clinical significance as the use of antimicrobial agents, intermittent positive pressure ventilation, expectorants and other supportive medical therapy allowed the
FIGURE 3. Microscopic (100 x) appearance of polypoid mass showing slight hyperplastic columnar epithelium and edematous loose areolar tissue with lymphocytic and plasma cell infiltrate.
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FIGURE 4. Fourth and final bronchial polyp, also bronchoscopically extirpated, on January 31, 1966. patient to recover completely and leave the hospital on February 9, 1966. During the past three years of follow-up, chest x-ray examinations have shown persistent opacification of the left lower lobe, reflecting either a chronic pleural reaction or possibly segmental atelectasis. In spite of this, antibiotics, branchodilators and expectorants have not been needed and there has been no recurrence of wheezing or "asthma." In fact since her discharge from the hospital, the patient has yet to experience pulmonary symptoms. DISCUSSION
The pathologic finding of polyps in the bronchial tree is most unusual. Patterson! in his review in 1930 could find only five such cases in the medical literature, but since then, there have been sufficient reports which indicate that bronchial polyps are more common than previously appreciated. Notwithstanding, the finding of multiple polyps in the distal tracheobronchial tree is indeed rare, most case reports describing a single or isolated polypoid lesion in the more proximal portions of the upper respiratory tract, ie, in the trachea and upper bronchi. Fibroepithelial polyps, unlike other polypoid bronchial tumors, do not exhibit the squamous epithelial over-growth of papillomas nor the bizarre and varied patterns of bronchial adenomas," Instead, they consist of a loose connective tissue core covered by normal bronchial respiratory epithelium, the core resembling the myxomatous regions of edematous nasal polyps." Unlike nasal polyps, however, fibroepithelial polyps are not usually multiple and are not necessarily found only in atopic individuals. In addition, they do not usually contain eosinophils. One notable exception, however, is the recent report of a patient with longterm bronchial asthma who did indeed have an eosinophil rather than a sparse plasma cell-lymphocyte infiltrated bronchial polyp."
282 Our patient was also thought to have bronchial asthma, and she, in fact, was treated for this condition for ten years prior to hospitalization, but none of her polyps contained eosinophils, and she never suffered from nasal allergy nor did she ever have nasal polyps. She also failed to have a noteworthy family history of atopy, and thus far, during the past three years since her discharge from the hospital, there has not been any recurrence of wheezing. Thus, the diagnosis of bronchial asthma appeared to be an error, and the bouts of wheezing were probably on the basis of recurrent transient episodes of polyp induced stop-valve type bronchial obstruction.v" The pathogenesis of bronchial polyps is still unresolved, and it is not certain whether the inflammation is a primary event or a secondary result of obstruction.t-" Thus bronchial polyps may be categorized as either inflammatory or neoplastic (inflammatory-like). In our patient, the bronchial polyps appear to be secondary to obstruction, since wheezing had been present for a number of years prior to the onset of bronchial inflammation, and endoscopic extirpation of the obstruction resulted in complete cure without the need for further use of antimicrobial agents. Although there was no clear-cut evidence here that the polyp was the end result of chronic bronchial inflammatory disease, the existence of such polypoid chronic bronchitis is well documented.t-v'? One such case was reported a few years ago and concerned a 34-year-old woman who had a history of chronic bronchitis since childhood.!" She developed bronchial obstruction due to heaped up polypoid granular tissue in the lumen of the left lower lobe bronchus. Although bronchoscopic removal of the offending tissue relieved her, she was not considered cured in view of continued bronchitis, thus anticipating future recurrence. In the same report, a second case was presented involving a 62-year-old laborer in whom there was a three to four week history of dyspnea, cough, yellowgreen sputum expectoration, and right-sided plural effusion. Bronchoscopy revealed a 2 ern granulomatous mass obstructing the right lower lobe bronchus. Endoscopic removal resulted in complete cure of the patient, and pathologic study revealed that the mass was actually a polyp clothed by columnar ciliated epithelium. The polyp's core consisted of edematous fibrous tissue which was vascular and which also contained acute inflammatory infiltrate. In spite of the pathologic findings reported in this man, the histology of our patient's bronchial polyps and of those reported by others failed to show acute inflammatory cellular or eosinophilic invasion.
KAHN AND AMER
Rather, sparse numbers of lymphocytes and plasma cells have predominated in almost all cases. Von Eicken, in 1907, was the first to use a bronchoscope for the diagnosis and removal of a benign bronchial neoplasm; and in 1910 Speiss was the first to report the bronchoscopic removal of a fibrous polypoid tumor. n- Ia Since then there have been numerous reports in the literature describing complete cure of bronchial tumors by means of bronchoscopic extirpation.1,4·11.14-111 It is true that the advent of improved surgical techniques has also resulted in the successful elimination of benign bronchial tumors by means of pulmonary resection and bronchotomy.Y-!? However, such improved techniques and successful results do not refute the statement of Jackson and Jackson,20 in 1938, that "almost all benign endobronchial tumors and tumor-like formations can be removed bronchoscopically and the prognosis as to life in such cases is good". The present case report reinforces the statement and shows that the use of endoscopic techniques still deserves serious consideration by the thoracic surgeon. A three year follow-up of our patient confirms the apparent success of bronchoscopic extirpation of the offending obstruction, there being a return of normal pulmonary drainage and a restoration of the patient to good pulmonary health. The clinical significance of the present case, therefore, is three-fold. First, it is obvious that fibroepithelial polyps of the bronchus can be mistaken clinically for carcinoma and lead to serious therapeutic errors. Second, although these lesions are benign both in growth and microscopic anatomic appearance, significant obstruction may result in impaired drainage of bronchial secretions producing secondary infection and a respiratory symptom complex. Third, the operative procedure required for the removal of these lesions is relatively simple with few hazards involved, and striking relief of the respiratory symptom complex can result. REFERENCES
1 2 3
4 5
PATTERSON, E.G.: Benign bronchial neoplasms, Arch. Otolarung., 12:739, 1930. ROWLANDS, D.T., JR.: Fibroepithelial polyps of the bronchus: A case report and review of the literature, Dis. Chest, 37:199, 1960. CALDAROLA, V.T., HARRISON, E.G., JR., CLAGETT, O.T., AND SCHMIDT, H.\V.: Benign tumors and tumor-like conditions of the trachea and bronchi, Ann. Otolaryng., 73:1042, 1964. PERONI, A.: Inflammatory tumors of the bronchi, Arch. Otolaryng., 19:1, 1934. REISNER, D.: A case of intrabronchial polypoid adenoma, with a review of the literature, Arch. Surgery, 16:1201, 1928.
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FATAL SUBDURAL HEMATOMA 6 SAINI, V.K. AND WAKE, P.L.: Inflammatory polyp of the bronchus, Ann. Thor. Surg., 5:141, 1968. 7 SALEK, J., PAZDERKA, S., AND ZAK, F.: Solitary bronchial polyps of inflammatory origin: A report of 2 cases treated by operation, J. Thor. Surg., 35:807, 1958. 8 CITRONI, G.A., AND DI GUGLIELMO, L.: Inflammatory polypi of the bronchi, Minerva Med., 46:686,1955. 9 JACKSON, C., AND JACKSON, C.L.: Benign tumors of the trachea and bronchi, JAMA, 99:1747,1932. 10 ASHLEY, D.J.B., DANINO, E.A., AND DAVIES, H.D.: Bronchial polyps, Thorax, 18:45, 1963. 11 MYERSON, M.C.: Benign neoplasms of the bronchus: Report of a case of fibrolipoma of the left main bronchus removed through the hronchoscope, Amer. J. M. Sci., 176:720, 1928. 12 VON EIKEN, c.. Bronchoscopische mitteilungen, Verhandl. d. Vereim Sud. Dent. Laryngol., 1907,410. 13 SPEISS, G.: Eine fall von hochgraduger dyspnoe infolge eines polypen in r. bronchus, Munchen Med. Wchmchr., 1910, 4, 2095. 14 JESBERG, S., Bronchoscopic studies of some conditions of the respiratory tract, Ann. Otolaryng., 35:509, 1926. 15 LEMANN, I.I., Bronchial polyp associated with mediastinal mass, Med. Clin. of N.A., 12:1329, 1929. 16 LINDGREN, A.G.H.: Benignant polypous bronchial tumors, Acta. Otolaryng., 27:183, 1939. 17 PELEG, J., AND PAUZNER, Y.: Benign tumors of the lung, Dis. Chest, 47:179,1965. 18 YANKAUER, S.: Benign tumor of bronchus, Laryngoscope, 39:549, 1929. 19 ZAMORA, A.M.: Two benign growths removed by bronchoscopy, J. Laryng. and Otol. 46:829,1931. 20 JACKSON, C., AND JACKSON, C.L.: Benign tumors and tumor-like conditions in the tracheobronchial tree, Amer. J. Surg., 42:275, 1938. Reprint requests: Dr. Kahn, 1128 Medical Drive, Tyler, Texas 75701.
Fatal Subdural Hematoma: An Unusual Complication of Pacemaker Failure* Alfred Heilbrunn, M.D. O O
C6nical pacemaker failures are usually manifested by return of complete heart block, recurrence of StokesAdams attacks or sudden death. In the case presented, the patient sustained cerebral trauma during a StokesAdams attack, but symptoms of his subdural hematoma did not become evident until five days after re-establishment of pacing. The posslbDity of intracranial trauma should be considered fonowing Stokes-Adams attacks especially if changes in cerebral status occur.
pacemaker systems are generally acI mplantable cepted as definitive treatment for patients with symptomatic complete heart block.':" However, the
°From the Department of Surgery, Veterans Administration Hospital, Kansas City, Missouri and the University of Kansas School of Medicine, Kansas City, Kansas. ooChief, Surgical Service and Associate Professor of Surgery.
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unpredictability of battery life remains a problem. Opinion regarding replacement has varied considerably.':" While Chardack! recommends elective replacement at 30 months, Zo1l6 recommends replacement only because of malfunction or evidence of battery exhaustion. The following patient is of interest since he developed a previously unreported complication of pacemaker failure after 43 months of continuous pacing. CASE REPORT
An 82-year-old white man was admitted to the Kansas City Veterans Administration Hospital in October, 1967 with syncopal episodes due to pacemaker failure. The patient had transient complete heart block in 1955. Following the onset of Stokes-Adams attacks in October, 1962, a pacemaker with myocardial electrodes was inserted at another hospital. Failure of this pacemaker occurred in February, 1964 and it was replaced at our hospital by an Electrodyne pacemaker with myocardial electrodes. The patient was seen at regular intervals with pacing at 72 per minute and asymptomatic. In October, 1969, syncopal episodes recurred and the patient was returned to the hospital the following day. The pulse rate was 28 per minute. Pacing was re-established using a temporary transvenous catheter electrode. Because of his advanced age (82), it was felt inadvisable to attempt another thoracotomy. A Medtronic implantable transvenous pacemaker was obtained for insertion. While discussing the proposed procedure with the patient he complained of pain in the hip which had been aggravated during insertion of the temporary catheter electrode. Therefore, following implantation of the permanent pacemaker system, x-ray films of the hips were obtained. but no evidence of bony injury was seen. For the first five days following the implantation of the new unit, the patient did well, pacing at 73 beats per minute without extrasystoles or arrhythmia. He was then noted to be increasingly lethargic, his fluid intake had become inadequate and his blood urea nitrogen had risen to 38 mg percent. However, in spite of correction of fluid and electrolyte problems, the patient's level of consciousness continued to deteriorate during the next 36 hours. No localizing signs were present. A spinal tap showed xanthochromic fluid and the possibility of subdural hematoma was then considered. Bilateral carotid arteriography was undertaken and demonstrated hilateral space-occupying lesions. Following neurosurgical consultation, hilateral frontoparietal trephinations with evacuation of subdural hematoma from both sides was accomplished. Postoperatively, the patient showed some improvement, but never regained consciousness. His condition gradually deteriorated, and in spite of intensive supportive management, he developed pneumonia and expired in January, 1968. Throughout the prolonged postoperative course, there was no difficulty with the cardiac pacemaker system. Autopsy examination showed resolving bilateral subdural hematomas. There were old infarcts in both occipital lohes of the cerebrum and acute confluent bronchopneumonia of the lungs. The transvenous pacemaker electrode was in good position in the apex of the right ventricle and throughout most of its course had become covered with the smooth thin layer of tissue. At the point where the electrode traversed the tricuspid valve was an area of fibrous thickening.