saecrse RePORTS Successful Treatment of Pleural Aspergillosis and Bronchopleural Fistula· Charlotte R. Colp, M.D ., F.C.C.P. °°and William A. Cook, M.D., F.C.C.P.t
We report the successful treatment of a patient with a bronchopleural fistula and pleural aspergilloma. Treatment consisted of intrapleural instillation of amphotericin B and nystatin followed by creation of an Eloesser flap. to the common occurrence of the various Compared forms of bronchopulmonary aspergillosis, infection
of the pleura by Aspergillus species remains rare. It has been reported as a complication of primary Aspergillus pneumonia, and as a complication following lung resection for aspergilloma, tuberculosis, and lung cancer.vf Bronchopleural fistulae after operation have seemed to be the major predisposing factor in the latter patients. A group of patients have also been reported with bronchopleura fistulae secondary to tuberculosis, either active or inactivet -f or sarcoidosis" who subsequently developed pleural infection with Aspergillus. Treatment with intrapleural instillation of antifungal agents has been recommended, and has led to eradication of the fungus in four of five cases." However, longterm foHowup of these patients had not been reported. We present the results of successful treatment of such a patient, with intrapleural administration of amphotericin B and nystatin followed by creation of an Eloesser flap for longterm drainage.
FIGURE 1. X-ray film of July, 1967 shows right-sided pleural thickening due to therapeutic pneumothorax, as well as inactive pulmonary tuberculosis. only, and 1-3 percent on all other occasions. Four specimens each of sputum and pleural fluid were negative for MIIObacterium tuberculosis on smear and culture. However, three specimens of pleural fluid, but no sputa, yielded a heavy growth of Aspergillus [umigatus, and the serum percipitin test against A fumigatus was positive . On admission to the hospital, thoracentesis was performed in the right posterolateral portion of the chest, and thiok greenish fluid was obtained. Thoracic surgical consultation _"J!l!""'_-~
The patient was a 56-year-old white woman who contracted pulmonary tuberculosis in 1942. She was treated with right-sided pneumothorax therapy for a right upper lobe cavity, with inactivation of the disease and cavity closure . From 1963 to 1966 isoniazid was given on a prophylactic basis. Chest x-ray films taken in 1967 show considerable right pleural thickening and some fibrosis in the lung, consistent with a history of inactive tuberculosis (Fig 1) . The patient remained well, with only occasional slight cough and sputum production, until March of 1971 when these symptoms worsened, and slight hemoptysis recurred. Chest x-ray films at that time showed a halo sign in the previous pleural density and a fluid level in the right lower portion of the chest, located in a posterior pleural pocket (Fig 2) . She was afebrile. Repeated white blood cell differential count~ showed 5 percent eosinophils on one occasion °From the Department of Thoracic Medicine and Surgery, Albert Einstein College of Medicine, Bronx, N.Y. ° °Associate Clinical Professor of Medicine. t Associate Professor of Surgery. Reprint requests: Dr. Colp, Room 612 Van Etten, Albert Einstein College of Medicine , Bronr 10461
96 COLP, COOK
, FIGURE 2. In 1971 fluid level is seen in right lower portion of chest, as well as halo sign in previous right ap ical pleural density.
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FIGURE 3. Injection of contrast material through chest tube results in filling of pleural pocket, of "cavity" containing fungus ball, and of bronchiectatic bronchi of right upper lobe. was obtained (WAC), and underwater tube drainage was begun. Ampicillin, isoniazid, and ethambutol were given. A persistent air leak was noted, confirming the presence of a bronchopleural fistula. A sinogram through the chest tube (Fig 3) showed contrast: material filling the pleural pocket, entering the cavity which contained an intracavity density (fungus ball), and right upper lobe bronchi, which appeared bronchiectatic. . When the nature of th~ infecting organism became'
FIGURE 4. December, 1973. Following antifungal treatments and longterm Eloesser drainage, fungous ball is no longer present at right apex. Fluid level has disappeared from right lower portion of chest, indicating healing of bronchopleural fistula.
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known, it was decided to treat' the patient with local irrigations of amphotericin B through the chest tube. Initially, 5 mg doses of amphotericin B were given, gradually increasing to 25 mg in each dose. This was diluted to a total volume of approximately 35 ml with 5 percent dextrose in water as specified by the manufacturer. We found that a larger volume of injected solution could not be accommodated by the pleural space and, regardless of the position in which the patient was placed, she would simply cough and expell the additional solution. These irrigations were given once daily, the solution being permitted to remain in the chest with the tube clamped and the patient recumbent or lying on the right side. After one hour the tube was undamped, .and the residual solution, mixed with pleural fluid, was permitted to drain out. The patient tolerated these treatments without sideeffect; there were no alterations in hemoglobin, hematocrit, blood urea nitrogen (BUN), or serum potassium, and the temperature rose to 38°C, on only ODe occasion. The patient never manifested any evidence of allergic aspergillosis. The amphotericin B instillations were continued from April 1 to April 30, with a total dosage of 750 mg. Within the first week the pleural fluid began to diminish in volume, and numerous bacteria, but not A fumigatus, were cultured. On April 22 and May 5 cultures of the pleuralefluid again grew A fumigatus. Sensitivity testing at that time indicated sensitivity to amphotericin B (at 25 I'g/ml) and nystatin (at 15 I'g/ml) . After correspondence with Dr. Krakowski of \yarsaw, we began nystatin installations of 30 ml containing 2,500 units/mL This was given daily for a month from May 14 to June 16, for a total dosage of 2,200,000 units. All cultures of pleural fluid were negative for fungus from May 9 on. Subsequently, the patient had complete disappearance of all symptoms, The air leak and some pleural drainage continued, but chest x-ray films showed disappearance of the fungus ball previously seen at the right apex. Thus, the patient still was left with empyema negative for A fumigatus and a bronchopleural fistula. The patient, with tertiary syphilis, a vital capacity 53 percent of predicted (only 60 percent of which was expired in the first second), and a lung scan showing decreased perfusion of the entire right lung and the left upper lung field, was not felt to be a candidate for right pleuropneumonectomy. Tube drainage seemed undesirable, Therefore, it was decided to create an Eloesser flap to facilitate longterm drainage of the right-sided empyema. t O This was performed on June 17, resecting portions of two ribs and suturing the superiorly based skin flap to the upper margin of the pleural defect, and was well tolerated. The patient left the hospital one week later and was able to care for the wound drainage with the help of her husband at home. The patient has had no recurrence of the fungus infection, nor, in fact, of any symptoms of significant infection in the chest. An Aspergillus precipitin test, one year later, was negative . Drainage of purulent material from the flap opening subsided gradually over a two-year period. Persistence of the bronchopleural fistula was apparent initially, with air blowing out of the flap when she coughed or strained, and a small persistent fluid level on, chest x-ray films. However, the patient noted that her air leak subsided completely during the first year, and after two years, chest x-ray 6lms showed that the air pocket was gone, and only a ~i£kene4 posterior pleura remained (Fig 4) . DISCUSSION
This patient had longstanding pleural thickening and possibly empyema due to a previous therapeutic pneu-
SUCCESSFUL TREATMENT OF PLEURAL ASPERGILLOSIS AND ASTULA 97
mothorax for pulmonary tuberculosis. In 1971 she developed a bronchopleural fistula, with Aspergillus infection of the pleura and the appearance of a "fungous ball." Bronchopleural fistulae as a late complication of therapeutic pneumothorax preceded Aspergillus infection of the pleura in all of the cases in which operation was not performed, as reported by Krakowski et al? and Voisin et al." The actual cause of the bronchopleural fistula is uncertain in our patient, since it was not associated with positive mycobacterial cultures; four of the patients reported by Voisin et al 8 also had no evidence of active tuberculosis. The development of a "pleural aspergilloma" has also been previously described. While in our patient it was difficult to determine radiologically whether the fungus ball was in a lung cavity or in the pleural space, the latter seems likely, for no lung cavity was seen in the chest x-ray film of 1967. Likewise in the other cases reported, Aspergillus infection of the pleura seemed to be a result of pleural disease rather than of rupture of a lung cavity into the pleura. All treatment in this case was intrapleural with no systemic therapy so that a parenchymallesion would not have been reached. In our patient, instillation of antifungal agents into the pleura led to apparently permanent eradication of the organisms and of the fungus ball. Since the bronchopleural fistula remained, and since the patient seemed a poor risk for major surgical repair, it was decided to create an Eloesser flap for longterm drainage of the pleural space. With this treatment, permanent closure of the bronchopleural flstula appears to have
1 Salfelder K, Capretti C, Hartung M, et al: Dos casos poco comunes de aspergilosis pulmonary pleural. Mycopathol Mycol AppI14:78, 1961 2 Barlow D: Aspergillosis complicating pulmonary tuberculosis. Proc R Soc Moo 47 :877, 1954 3 Monod 0, Dieudonne P, Tardieu P: Les aspergilloses pulmonaries postoperatoires. J Fr Moo Chir Thorae 18: 579,1964 4 Golebiowski AK: Pleural aspergillosis following resection for pulmonary tuberculosis. Tubercle 39: In, 1958 5 Sitkowski W, Piotrowski A, Kryszkiewicz T: Aspergillus fumigatus Iako przyczyna powiklania pooperacyjnego. ( AspergiUus fumigatus as the cause of postoperative complications). Gruzlica 27: 1069, 1959 6 Sochocky S: Infection of pneumonectomy space with AspergiUus fumigatus treated by nystatin. Dis Chest 36: 554, 1959 7 Krakowka P, Rowinska E, Halweg H: Infection of the pleura by Aspergillus fumigafus. Thorax 25 :245, 1970 8 Voisin C, Sergeant YH, Wallaert MC, et al: L'aspergillose pleurale ( Aspects etiologiques, Anatomo-cliniques et therapeutiques. A propos de 15 observations) . Rev Tuberc Pneumol33:477, 1969 9 Irani FA, Dolovieh J, Newhouse MT: Bronchopulmonary and pleural aspergillosis. Am Rev Resp Dis 103:552, 1971 10 Eloesser L: Of an operation for tuberculous empyema. Ann Thorac Surg 8:355, 1969
98 MAULL, Me ELVEIN
Infarcted cExtralobar Sequestratien · •
Kimball I. Matdl, M.D.,·· and Richard B. McEloein, "M.D.t
A 32-year-old man who presented with symptoms 8111gestiDI empyema was foud at thoracotomy to have Infarction of an eDndo.... pulmonary sequestration. He w. completely reUeved by esdsion of the sequestration after securiDg the systemic vascular pedicle which bad undergone torsion. Recopition of the BDOmalo.. bloody supply and other colllenital anomalies In such cases Is emphasized.
ulmonary sequestration is a manifestation of disturbed embryonic development with nonfunctioning lung tissue receiving its vascular supply from the systemic circulation. Two forms of pulmonary sequestration are described, intralobar and extralobar. Infarcted pulmonary sequestration of either type has not been described previously. This is a report of a patient with an infarcted extralobar pulmonary sequestration, an unusual complication of an uncommon entity.
CASE REPoRT A 32-year-old white man presented with a two-week history of intermittent, severe, pleuritic, nonradiating anterior pain in the left side of his chest. His initial treatment with analgesics was ineffective, and a chest x-ray film 11 days prior to admission showed a left lower-lobe infiltrate and pleural effusion. The patient was started on therapy with antibiotics and expectorants when he developed fever and chills. No therapeutic response occurred. His pain grew more severe, and the patient had increasing dyspnea, insomnia, and fatigability. His past history was unremarkable. Physical examination revealed an acutely ill and mildly tachypneic man complaining of pain in the left side of his chest. The thorax was of normal configuration with dullness to percussion over the lower left portion of the thorax. Breath sounds were absent in this same area, with diminished breath sounds superiorly. The right side of the thorax was normal. Laboratory data on admission included a hematocrit reading of 39 percent and a leukocyte count of 12,BOO/cu mm with 71 percent segmented neutrophils and 5 percent stab forms. Chest roentgenogram showed what was thought to be an enlarged left pleural effusion. Thoracocentesis was perfonned, but no fluid or purulent material was obtained. On the second hospital day, left thoracotomy was performed. At operation, BOO ml of slightly serosanguineous sterile fluid was removed. A thickened fibrinous peel was identified over the atelectatic left lower lobe, diaphragm, and chest wall. A discrete mass of purplish-red firm tissue was found posterolateral to the left lower lobe. It had a pleural covering and a single pedicle which had twisted upon itseH. The ·From the Department of Surgery, Good Samaritan Hospital, and the University of Kentucky Medical Center, Lexington. Resident/Surgery, University of Kentu cky Medieal ··Chief 1 Center. tclinical Associate Professor of Surgery, University of Kentucky Medical Center. Reprint reqtle8t8: Dr. McElvein, 2121 Nicholasville Rd., lexington, Kentuckf/40503
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