Early Post-Pneumonectomy Bronchial Stump Fistula

Early Post-Pneumonectomy Bronchial Stump Fistula* Arthur II . Pomerantz , M.D ., Ph.D .; Manjul D. Derasari , M.D .; Surinder S. Sethi , M.D .; and Saleem Khan , M.D .

Bronchial stump fistula occurred on the seventh postoperative day in a 45-year-old man who underwent right pneumonectomy for T2N2MO adenocarcinoma of the lung. It was attributable to ventilator-related barotrauma during treatment of ARDS from multiple pulmonary emboli. A frequently lethal complication, its presentation, management and prevention in the early postoperative period are discussed.

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ost-pneumonectomy bronchopleural fistula is a recognized complication of aggressive loco-regional treatment of carcinoma of the lung, occurring not infrequently when pulmonary resection follows external radiation therapy. I Its median incidence worldwide is between 7 and 9 percent.' Most fistulae present no sooner than four to six weeks postoperatively and usuall y result from an empyema of the post-pneumonectomy space ," Herein we present an unusual case of early post-pneumonectomy bronchial stump fistula without antecedent empyema or radiation therapy. Unlike late post-pneumonectomy fistulae, it occurred prior to mediastinal fixation, initiall y pre senting as a tension pneumothorax with direct compromise of remaining contralateral pulmonary ventilation. CASE REPORT

A 45-year-old longshoreman with a histor y of heavy cigarette smoking and asbestos exposure presented to his family physician with episodic bilateral leg and th igh edema and was found to have an asymptomatic, 4 cm right upper lobe mass with likely hilar adenopathy (Fig 1). After bilateral lower extremity venography showed no evident deep venous thrombosis, he was referred for bronchoscopic examination which documented adenocarcinoma of the lung. After outpatient metastati c work-up, including head and abdominal cr scanning and bone scanning revealed no extrathoracic disease and cr scanning showed no evident mediastinal adenopathy, he underwent right thoracotom y with pneumonectomy and mediastinal node dissection for clinical T2Nl disease . His preoperative FEV 1 was 3.1 L with normal room air arterial blood gas levels, and there was no hemod ynamic instability intraoperatively during trial digital occlusion of the right pulmonary artery. At surgery, there was extracapsular tumor involvement of superior mediastinal lymph node , levels 2-4 requiring en bloc resect ion of pericardium along the ascending aorta for clearance . The level 11 hilar lymph nodes and level 7 subcarinal nodes showed subcapsular tumor involvement, but all resection margins were tumor-free microscopically, and resection was deemed complete. A standard, mechanically stapled bronch ial closure was performed 1 cm distal to the carina. Antibiotic prophylaxis included perioperative intravenous cefazolin and intraoperative topical cefazolin. The immediate postoperative course was unremarkable with the patient extubated in the recovery room and out of bed tolerating oral liquids on the first postoperative day. On the evening of the second postoperative day, while ambu *From the Department of Surgery Division of Thoracic Surgery, Albert Einstein Medical Center, Mount Sinai-Daroff Division, Philadelphia. Reprint requests : Dr. Pomerantz , 308 Lombard Street , Philadelphia 19147 654

FIGURE 1. Preoperative chest roentgenogram . Right hilar adenopathy is suggested , consistent with clinically staged T2NIMO adenocarcinoma . lating on the floor, he developed acute dyspnea and tachypnea and was found to be hYPOXic with respiratory alkalosis, in atrial flutter with a ventricular respon se rate of 160. After bolus anticoagulation with heparin, he was transferred to the multid isciplinary ICU for invasive hemodynam ic monitoring, including continual blood gas determinations. He was intubated orotracheally the next morning for progress ive ARDS unresponsive to furosemide diuresis or chemical conver sion of supraventricular tachycardia with verapamil and digoxin (Fig 2). His oxygenation improved steadily with controlled positive pressure ventilation with Flo. 1.0 and 12.5 cm PEEP and he was weaned to an Flo. of 0.6 on postoperative day 6.

FIGURE 2. Chest roentgenogram three days after right pneumonectomy for pathologically staged T2N2 disease . Note the ARDS pattern of infiltration of the remaining lung. The right hemithorax has not yet filled completely with fluid. Post-pneumonectomy Bronchial Slump Fistula (Pomerantz et aJ)

FIGllRE 4. Chest roentgenogram demonstrates acute management of the bronchial stump fistula with selective, left main bronchus intubation and tube thoracostomy with water seal drainage of the right hemithorax. Note correction of the mediastinal shift compared to Figure 3. increased right pneumothorax, increasing air leak via the Pleurevac, and leftward mediastinal shift requiring reintubation of the left main bronchus. On the 13th day post-pneumonectomy, again on a T-piece, he underwent transjugular placement of a Greenfield inferior vena caval filter, and the next day, was returned to the operating room off heparin therapy for repeat right thoracotomy debridement and reclosure of the right bronchial stump. At surgery, air leak was detected at the caphalad end of the staple line, which was excised . The right bronchial origin was closed at the carina with interrupted silk sutures and reinforced with a posteriorly based intercostal muscle flap. The closure was tested to 55 em H,O of static pressure without air leak. Operative fluid cultures grew alpha streptococci and Candida sp, necessitating change of antibiotics to FIGURE 3. Chest roentgenograms before (upper) and after (lower) development of the bronchial stump fistula while on positive pressure ventilation on the seventh postoperative day. Note complete reversal of right hemithorax opacification with subcutaneous emphysema and mediastinal shift after fistula formation. His continuous IV heparin requirement throughout remained 3,000

Ulh for adequate anticoagulation (PTT 2 x control).

On the morning of the seventh postoperative day, right-sided subcutaneous emphysema was noted, together with acute hypercarbia to Pco, of 60 mm Hg, associated with a chest x-ray film change (Fig 3). Upon review of the ventilator record , an intermittent sigh cycle had been added the evening before as part of a "routine protocol" with sigh volumes of 1,400 rnl, twice the tidal volume, and measured peak sigh pressures of 65-70 cm H,O The bronchial stump fistula was managed acutely with right tube thoracostomy and waterseal drainage offsuction combined with left main bronchus intubation with a No 8 cuffed orotracheal tube using the fiberoptic bronchoscope (Fig 4). Inspection of the right bronchial stump disclosed no evident gaps in the staple line. Antibiotic prophylaxis and IV cefoxitin and gentamicin were begun at this time. although the pleural fluid was sterile to culture. Because of repeated proximal displacement of the orotracheal tube with resultant tidal air leak out the chest tube, it was replaced the next day with a left-sided Bronchocath double lumen tube. On the lIth postoperative day; the patient was successfully weaned to a T-piece with Flo, 0.4 and subsequently extubated. After 13 hours, tbe patient developed tachypnea with CO, retention to a Pco, level of 60 mm Hg,

FIGURE 5. Chest roentgenogram after operative reclosure of the fistula two weeks after pneumonectomy. Selective left main bronchus intubation is maintained with a double lumen catheter (Bronchocath). The chest tube has been clamped. CHEST I 93 I 3 I MARCH, 1988

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cefatoxime and ketoconazole. Over the subsequent week, the patient was maintained on controlled volume ventilation via the double lumen bronchial catheter, receiving intravenous hyperalimentation, with gradual resolution of a left lower lobe infiltrate but little weaning progress (Fig 5~ He was transferred to another facility for high-frequency jet ventilation and bronchial decannulation on the 21st day post-pneumonectomy He tolerated jet ventilation well, initially via an orotracheal tube and subsequently via tracheostomy with slow weaning improvement and without fistula recurrence. Unfortunately in the early morning of the 26th postoperative da~ he suffered acute respiratory arrest from apparent tracheal occlu~ionl tracheostomy tube dislodgement. He was returned to positive pressure ventilation and developed recurrence of the bronchial fistula 12 hours later. He died on the 28th day post-pneumonectomy with worsening left lower lobe pneumonia and staphylococcal sepsis. DISCUSSION

Clearly this case represents a very unlucky individual, stricken with locally advanced cancer, in the fifth decade of life, facing radical surgery as the only hope for cure, and having survived that immediate insult, unrelentingly smitten with repeated, catastrophic complications to which he eventually succumbed. Nevertheless, it presents a particularly graphic example of an uncommon thoracic surgical problem, post-pneumonectomy bronchial fistula occurring within the first postoperative month. As seen in Figure 3, this case exaggerates the pathognomonic x-ray film finding of postpneumonectomy stump fistula: namely a lowering of the air/ fluid level in the pneumonectomy cavity due to fluid loss through the fistula into the trachea and contralateral bronchus. Herein, because of positive pressure ventilation at the time of occurrence, there is no residual fluid left in the pneumonectomized space. In addition, subcutaneous emphysema is particularly apparent. Because of intubation and sedation, the classic presentation of abrupt onset of persistent, hacking cough productive of copious, bloody fluid was not seen in this patient. 3 Unlike late post-pneumonectomy bronchopleural fistulae which arise from infection of the fixed, fibrotic post-pneumonectomy space, early postoperative fistulae, as in this case, are further complicated by a mobile mediastinum whose contralateral shift readily compromises remaining lung function. Whereas tube thoracostomy or open drainage of the post-pneumonectomy space are all that is needed acutely in the management of late fistula and empyema by preventing spill-over of infected fluid into the remaining lung, additional control of the bronchial stump air leak is essential for early fistula in order to prevent recurrent tension pneumothorax and contralateral mediastinal displacement. In particular; with major bronchial fistula, the option of constant chest tube suction is eliminated, as this serves only to asphyxiate the patient. In this case the problem of early management of bronchial stump air leak was further compounded by the immediate need for continued positive pressure ventilation with PEEP in order to counteract ARDS in a poorly compliant remaining left lung. Herein, initial control of the air leak was readily achieved by selective left main bronchus intubation, initially with an orotracheal tube and subsequently with a double lumen tracheobronchial catheter, thus isolating the right bronchial stump from air flow This temporizing measure allowed enough resolution of remaining left lung compromise to permit weaning off 858

positive pressure ventilation. Unfortunately even at two weeks postoperation, mediastinal mobility persisted in this patient with gradual recurrence of ventilatory compromise with mediastinal shift after 13 hours of extubation, necessitating selective bronchial reintubation and subsequent reoperation for definitive surgical repair of the bronchial stump leak. Anecdotally it should be noted that at no time could the bronchial stump dehiscence actually be visualized at bronchoscopy The bronchial stump staple line always appeared intact on endobronchial inspection. In this particular patient, reoperation for surgical closure of the fistula was mandated as soon as remaining lung function permitted in order to remedy ventilatory failure and recurrent tension pneumothorax after extubation. For late post-pneumonectomy fistula, operative closure with stump revision has also been advocated on an elective basis in order to prevent ongoing soilage of the post-pneumonectomy cavity with oropharyngeal flora.2,4 The transmediastinal approach to the long, leaking bronchial stump through a previously unviolated, sterile field has been shown effective by some, though the right thoracotomy approach is more generally applicable and provides better exposure of the carina and both proximal mainstem bronchi.s" Long-term success rates for reclosure of operative fistula are definitely improved with pedicled muscle flap reinforcement which is felt to provide needed revascularization for stump healing, but still average only 50-70 percent in centers with a large experience.v' Fistulae do tend to recur after reclosure most of the time in the face of culture-positive, ongoing pleural infection, as reported herein." Nevertheless, reoperative stump closure usually buys enough time prior to fistula recurrence to allow mediastinal fixation to occur and thus greatly simplifies management to an empyema situation amenable to open drainage alone. In retrospect, this patient would have best been served with early high-frequency jet ventilation, ideally at the time of initial intubation for his pulmonary embolism-related ARDS. Completely compatible with PEEP therapy highfrequency jet ventilation assures minimal static airway pressures not significantly different from therapeutic PEEP pressures and would no doubt have eliminated the volume ventilator-associated barotrauma which is a likely contributor to early post-pneumonectomy Bstula." Unfortunately jet ventilation equipment still is not readily available even at major thoracic surgical centers, despite its relatively simple, reliable and low-cost technology Although the nationwide incidence of post-pneumonectomy bronchial stump fistula has recently been quoted as only 2 percent in the United States, it remains a significant proximate cause of postoperative mortality 2 We suspect that its incidence in the early postoperative period would be lowered by the prophylactic use of the high-frequency jet ventilator for all patients requiring prolonged mechanical ventilation after pulmonary resection. REFERENCES

1 Wilkins EW Jr. Bronchopleural fistula: prophylaxis. In: Grillo He, Eschapasse H, eds. International trends in general thoracic surgery vol 2, Major challenges. Philadelphia: W. B. Saunders, 1987:394-97 2 Bruni F: Bronchopleural fistula: treatment of the long stump after Post-pneumonectomy Bronchial Stump Fistula(Pomerantz et 8/)

pneumonectomy. Ibid, pp 413-21 3 Lams P. Radiographic signs in post pneumonectomy bronchopleural fistula. J Can Assoc RadioI1980 ; 31:178-80 4 Brewer LA III . Bronchopleural fistula: management. In: Grillo HC, Eschapasse H, eds. International trends in general thoracic surgery. vol 2, Major challenges. Philadelphia: W.B. Saunders, 1987:398-406 5 Perelman MI, Rymko LP, Ambatiello GP. Bronchopleural fistula: surgery after pneumonectomy. Ibid, pp 407-12 6 Williams NS, Lewis CT. Bronchopleural fistula: A review of B6 cases. Br J Surg 1976; 63:520-52 7 Fischer HBJ, Smith BE. High-frequency ventilation. In: Grillo HC, Eschapasse H, eds. International trends in general thoracic surgery. Vol 2, Major challenges. Philadelphia: \V.B. Saunders, 1987:207-16

Thoracic Hemangiomatosis Masquerading as Interstitial Lung Disease* jomes R. Vevaina, M.D ., F.C.C.P.; and Eugene]. Mark, M.D .

Pulmonary capillary hemangiomatosis is an extremely rare condition, first described by Wagenvoort. In this condition, sheets of thin-walled blood vessels infiltrate the lung parenchyma, bronchioles, and pleura. Three previous cases have been described in the literature. We describe a fourth. It probably represents a congenital abnormality. *From the Division of Pulmonary and Critical Care Medicine, Department ofInternal Medicine, Flushing Hospital and Medical Center, Flushing, NY; and the Department of Pathology, Massachusetts General Hospital. Harvard Medical School. Boston. Reprint requests: Dr: Vevaina, 45-18 Parsons Blvd, Flushing, New York 11355

horacic hemangiomatosis is an extremely rare condition T ofthe thorax of unknown etiology. In this condition, thin-

walled capillary blood vessels infiltrate the lung parenchyma, blood vessels, interlobular septa, bronchial walls, and the pleura and pericardium. It is not clear whether the condition is a neoplastic process or a congenital abnormality. The physiology of the condition is precapillary hypertension. A restrictive ventilatory pattern is seen on pulmonary function testing. Open lung biopsy may be diagnostic . Factor VIII staining of endothelial cells may be helpful in distinguishing a congenital abnormality from a neoplastic process. The clinical course is characterized by pulmonary hypertension and is inexorably downhill. It is impossible to differentiate from veno-oeclusive disease. Diagnosis poses considerable difficulty for the clinician and pathologist. CASE REPORT

A 25-year-old black woman with congenital scoliosis was admitted to the hospital with a diagnosis of interstitial lung disease and a moderate pericardial effusion. She had been dyspneic since age 13 and had a history of repeated episodes of respiratory infections. She also complained of cough and shortness of breath. There was no hemoptysis . A week prior to admission, dyspnea became worse and progressive. She also complained of faintness . She was started on therapy with bronchodilators. During a previous admission in another hospital, she was diagnosed as having nonspecific pulmonary fibrosis on the basis of an open lung biopsy. Kveim skin test result was reported as normal . On admission, the patient was severely orthopneic and complained of a choking sensation upon lying down. The vital signs were : pulse rate, 120/min; blood pressure, 140/90 mm Hg; temperature, 37.8°C; and respirations, 28/min. Physical examination of the chest revealed a silent heart, decreased breath sounds and diffuse fine rales. Peripheral edema was noted . Achest x-ray film (Fig 1)showed widening of the cardiac and mediastinal silhouettes and loss of the normal cardiac contours . Interstitial reticulonodular infiltmtions were present bilaterally from apex to

FIGURE 1. Chest x-ray film shows marked widening of the cardiac and mediastinal silhouette. There is a diffuse bilateral reticular-nodular infiltrate ,

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