Pancreaticobronchial Fistula

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1 GASTROENTEROLOGY 1986;90: Pancreaticobronchial Fistula Case Report and Review of the Literature J. DIRK IGLEHART, CHARLES MANSBACK, RAYMOND POSTLETHWAIT, LEROY ROBERTS, Jr., and WAYNE RUTH Duke University Medical Center, Veteran's Administration Medical Center, Durham, North Carolina Fistulas between the pancreas and bronchial tree are uncommon examples of internal pancreatic fistulas. We describe a case of pancreaticobronchial fistula in which the diagnosis was made by bronchoscopy and biochemical analysis of respiratory secretions. Endoscopic retrograde cholangiopancreatography was confirmatory and guided treatment by internal drainage of the pancreatic duct. Ten other cases were found in the Western literature that emphasize the principles of diagnosis and treatment. Internal pancreatic fistulas are well-recognized complications of acute and chronic pancreatitis and pancreatic trauma. These fistulas originate from a disruption in a major pancreatic duct, allowing the escape of exocrine secretions. A communication with the peritoneal cavity may result in pancreatic ascites. If the secretions are contained in the retroperitoneum, they can burrow upwards along the diaphragmatic crura and escape into the thorax. The thoracic presentation of internal pancreatic fistulas includes unilateral or bilateral pleural effusions, mediastinal pseudocysts, and fistulas into the tracheobronchial tree. Communications between the pancreas and the bronchial tree are rare and unusual examples of internal pancreatic fistulas. We have found only 10 cases of pancreaticobronchial fistulas reported in the American and Western European medical literature (1-9). In this study, we report an additional case in which the diagnosis was suggested by biochemical analysis of bronchial fluid and confirmed by endoscopic retrograde cholangiopancreatography (ERCP). This strategy followed the principles outlined for the Received March 18, Accepted September 17,1985. Address requests for reprints to: J. Dirk Iglehart, M.D., Box 3873, Duke University Medical Center, Durham, North Carolina by the American Gastroenterological Association /86/$3.50 investigation of internal pancreatic fistula in general (10-12). Our patient was successfully treated by internal drainage of the pancreatic ductal system using a Roux-en-Y loop of jejunum. Case Report The patient is a 66-yr-old black man whose current illness began in February 1983, when he presented with bilateral pleural effusions and complained of shortness of breath and a cough productive of copious frothy white sputum. He denied abdominal pain, nausea, or vomiting. There was a history of chronic and heavy alcohol consumption until 2 yr before this illness. The abdomen was flat with no fluid demonstrable, no tenderness, and no masses. Blood counts and electrolytes were normal, and room air arterial blood gas determinations revealed mild hypoxemia. His serum amylase was not determined. Liverassociated enzymes, calcium, protein, and albumin were all normal. A chest x-ray revealed a large right and a smaller left pleural effusion. Thoracentesis revealed serosanguineous fluid, although an amylase determination was not done. Computed tomography of the chest was normal, and abdominal computed tomography images revealed calcifications in a normal-sized pancreas. Ultimately, a right exploratory thoracotomy and lung biopsy failed to show any abnormalities. The patient was discharged in March 1983 with small bilateral pleural effusions. The patient was well until May 1983, when a chest x-ray showed a recurrent large right and minimal left pleural effusion. Thoracentesis of the right pleural cavity returned >1 L of serosanguineous fluid. The amylase content of this fluid was 1425 IU/L. Subsequent thoracentesis of the left pleural cavity returned serosanguineous fluid with an amylase content of 1923 IU/L. He required needle drainage of the right and left pleural cavities on subsequent occasions, and amylase contents in the fluids were consistently Abbreviation used in this paper: ERCP, endoscopic retrograde cholangiopancreatography.

760 IGLEHART ET AL. GASTROENTEROLOGY Vol. 90, No.3 Figure 1. The preoperative chest roentgenogram demonstrates a left lower lobe pneumonia with atelectasis.

Two separate attempts at ERCP were unsuccessful because of scarring of the duodenum from previous ulcer disease.

2 760 IGLEHART ET AL. GASTROENTEROLOGY Vol. 90, No.3 Figure 1. The preoperative chest roentgenogram demonstrates a left lower lobe pneumonia with atelectasis. At this time, minimal pleural effusions are present. >1000 IU/L. Multiple determinations of serum amylase were all normal. Two separate attempts at ERCP were unsuccessful because of scarring of the duodenum from previous ulcer disease. The patient was seen again in October 1983, with pleuritic chest pain and increased pleural effusions. He was treated for a left lower lobe pneumonia and returned later in the same month with the onset of shortness of breath and expectoration of large quantities of white frothy sputum, which was occasionally tinged with blood. Chest x-ray showed a left lower lobe pneumonia and pleural effusion (Figure 1). His sputum cultures demonstrated Enterobacter cloacae and he was treated with intravenous cefamandole. Initially, the patient improved; however, on the 20th hospital day, his condition worsened with tachypnea, left upper quadrant abdominal tenderness, and worsening of his left lower lobe pneumonia. A computed tomography scan of the abdomen now showed a retrogastric process consistent with pancreatitis and a peri pancreatic inflammatory reaction. The serum amylase was normal. The patient coughed large amounts of frothy clear sputum. Emergency bronchoscopy revealed moderately severe tracheobronchitis, and a large amount of frothy clear secretion came from the left lower lobe bronchus. An amylase determination on the aspirated secretions was >21,000 IU/L when diluted 1:24. A tentative diagnosis of pancreaticobronchial fistula was made, and the patient was intubated for control of his secretions. Repeat bronchoscopy revealed worsening of the acute bronchitis with edema around the left lower lobe orifice. Bronchial washings were collected separately from the right and left lower lobe bronchi. To assure the pancreatic origin of the enzyme activities, both amylase isoenzyme activity and trypsinlike immunoreactivity were determined (Table 1). Preoperatively, all of the amylase activity was in the pancreatic isoenzyme fraction, and trypsin immunoreactivity was present. A small step up in activity was present in the left bronchial tree. Endoscopic retrograde cholangiopancreatography was successfully performed and showed irregularity of the proximal portion of the pancreatic duct, with stenosis and extravasation of dye from the distal pancreatic duct, which tracked upwards toward the left crux of the diaphragm (Figure 2). One day later, the patient was taken to the operating room, and exploration showed a firm and woody pancreas. A dilated pancreatic duct was palpable. Superior to the pancreas there was marked induration of tissues adherent to the undersurface of the stomach, although no pseudocyst was present. The pancreatic duct was incised longitudinally and drained with a defunctionalized Roux-en-Y loop of jejunum (Puestow's procedure). The retrogastric space was drained externally. On the fifth postoperative day, the patient was extubated, and he made a gradual recovery with institution of feeding by the 10th postoperative day. Repeat bronchoscopy 3 wk postoperatively showed resolution of his bronchitis and a marked decrease in the amount of bronchial secretions. Lung secretions were again collected for biochemical analysis. Neither pancreatic amylase activity nor trypsinlike immunoreactivity was now detected (Table 1). The presence of salivary amylase activity is unexplained but could be due to contamination during passage of the bronchoscope through the pharynx. The patient has remained well, has gained weight, and has no cough. A chest x-ray 6 mo postoperatively was entirely normal (Figure 3). Discussion In our patient, an internal pancreatic fistula to the tracheobronchial tree was preceded by a history of alcohol abuse but without documented inflammatory disease of the pancreas. There was no abdominal pain until mild left upper quadrant discomfort was reported just before establishing the presence of amylase in bronchial secretions. Of 10 pancreaticobronchial fistulas reported in the Western literature, seven were preceded by acute fulminant pancreatitis (Table 2). Two additional cases followed violent crush injuries to the abdomen, both requiring surgical exploration, at which time severe left upper Table 1. Enzyme Activity in Bronchial Fluidso Preoper- Postoper- Enzyme Source ative ative Amylase (total) Both lungs THTC b 0 Amylase (pancreatic Right lung 12,500 c 0 fraction) Left lung 13,200 0 Amylase (salivary fraction) Right lung 0 3,000 Left lung Trypsinlike immuno- Right lung 2,500 d 0 reactivity Left lung 10,000 0 a Simultaneous serum amylase was always <75 lull. b Too high to count. C Units are international units per liter. d Units are nanograms per milliliter of trypsinlike immunoreactivity.

March 1986 PANCREATICOBRONCHIAL FISTULA 761 Figure 2. Endoscopic retrograde cholangiopancreatography (ERCP) following tube removal demonstrates opacification of the pancreatic duct (open arrows).

Extravasation of contrast into the retrogastric region was also demonstrated (arrow). Contrast was not observed to flow into the left thorax.

One case occurred in the setting of chronic relapsing pancreatitis (Table 2). The etiology of pancreatitis included alcohol, gallstones, and operative manipulation.

3 March 1986 PANCREATICOBRONCHIAL FISTULA 761 Figure 2. Endoscopic retrograde cholangiopancreatography (ERCP) following tube removal demonstrates opacification of the pancreatic duct (open arrows). Areas of pancreatic duct narrowing proximally and distally were demonstrated (arrowheads). Calcifications about the distal duct were also observed on computed tomography (not shown). Extravasation of contrast into the retrogastric region was also demonstrated (arrow). Contrast was not observed to flow into the left thorax. Opacification of the gallbladder (gb) and upper gastrointestinal tract is also demonstrated. quadrant injuries were documented (Table 2). One case occurred in the setting of chronic relapsing pancreatitis (Table 2). The etiology of pancreatitis included alcohol, gallstones, and operative manipulation. A cause for pancreatic disease was never determined in 5 reported c~ses (Table 2). It would apear that internal pancreatic fistulas may complicate acute or chronic pancreatic inflammatory disease of any etiology. Although the pancreatic disease underlying internal fistulas to the pleural or peritoneal cavities may be clinically silent (10), in a series of patients with pancreaticobronchial fistulas, acute fulminant pancreatitis frequently preceded the bronchial fistula. The presenting symptoms of pancreaticobronchial fistula direct attention away from the abdomen and toward the respiratory tree. Cough, dyspnea, and copious sputum production usually accompanies the appearance of the bronchial fistula (Table 2). The sputum produced is remarkable in both quantity and quality. In 9 of the 10 reported cases and in the present case, copious amounts of sputum are described. In our patient, this was a striking feature; hugh amounts of thin, "frothy" sputum were produced and required intubation to control. The term "frothy" is an apt description and appeared in the case histories of 4 patients from the literature (Table 2). Although the pancreatic enzymes present in the bronchial tree are not activated, they are irritating to the bronchial mucosa and, in our case, produced a severe tracheobronchitis. In one other case, described by Cox (8), bronchoscopy was done and marked bronchitis was observed. Hoarseness accompanied the tracheitis in our patient and was described in the case report of Sease (2). The diagnosis of pancreaticobronchial fistula was usually confirmed by demonstration of elevated levels of pancreatic enzymes in bronchial secretions or by injection of colored dye into external pancreatic fistulas and its appearance in coughed material (Table 2). In 6 reported cases, fistulograms were obtained by injection of radiopaque contrast media into external fistulas or into the pleural cavity. Our ~ase was the first to employ ERCP to help delineate a fistula from the pancreas to the bronchial tree. Endoscopic retrograde cholangiopancreatography is recommended to outline the anatomy of the fistula Figure 3. A postoperative chest roentgenogram demonstrates resolution of the left lower lobe pneumonia and no residual pleural effusion.

4 762 IGLEHART ET ill. GASTROENTEROLOGY Vol. 90, No.3 Table 2. Clinical Features of Pancreaticobronchial Fistula Reported in the Literature AuthorO Cause Symptoms and course Diagnosis Treatment Outcome Hunt (1) Trauma External fistula, cough, Sinogram, "ferments" in Distal pancreatectomy Cured frothy and copious spu- sputum tum, hemoptysis Hunt (1) Acute pancreatitis Pancreatic abscess, external Sinogram External drainage Cured (idiopathic) fistula, cough, copious and frothy sputum Sease (2) Acute pancreatitis External fistula, cough, co- Methylene blue in spu- Removal of stone im- Cured (gallstones) pious and frothy sputum, tum after injection in pacted in ampulla of hoarsllness sinus tract Vater, external drainage Ruffo et al. (3) ilcute pancreatitis Extllrnal fistula, left lung Amylase in lung abscess Abdominal and thoracic Cured (idiopathic) abscess, cough fluid drainage Dignan (4) Acute pancreatitis External fistula, cough, co- Sinogram and methyle!le External abdominal Cured (idiopathic) pious sputum, left lung blue in sputum after drainage followed by abscess sinus tract injection anastomosis of fistula to stomach Kaye (5) Acute pancreatitis Dyspnea, cough, copious Pancreatic enzymes in Tube thoracostomy Cured (idiopathic) and frothy sputum, large pleural fluid, broncholeft hydropneumothorax gram after contrast injection into pleural effusion Dubois et al. Acute pancreatitis Dyspnea, external fistula Sinogram via abdominal External drainage Cured (6) (idiopathic) fistula produces bronchogram Bell (7) Chronic pancreatjtis Previous cyst-jejunostomy Abdominal distention Supportive Died (alcoholic) for pancreatic pseudo- (meteorism) after posicyst, developed cough, tive pressure respiradyspnea and respiratory tion. Autopsy confailure firmed fistula Cox et a\. (8) Trauma Empyema and abdominal High sputum amylase Abdominal and thoracic Cured abscess after splenectomy drainage and repair of ruptured diaphragm Grossman et al. Acute pancreatitis Pneumonia and hydro- Methylene blue in spu- Drainage of empyema, Cured (9) (postoperative) pneumothprax after tum and contrast in subtotal pancreatectrans duodenal sphincter- pancreatic bed after in- tomy otomy and pancreatic jection in pleural needle biopsy cavity Current report Chronic pancreatitis Recurrent pneumonia and Pancreatic enzymes in Roux-en-Y pancreatico- Cured (alcoholic) pleural effusions, copious bronchial secretions, jejunostomy and frothy sputum ERCP ERCP, endoscopic retrograde cholangiopancreatography. a Numbers in parentheses are reference numbers. and pancreatic ductal pathology. The usefulness of ERCP in the diagnosis of internal pancreatic fistulas has been emphasized by several authors (10-12). Pertinent information obtained includes location of the fistula, the size of the p;mcreatic duct, the presence and location of ductal strictures, and the presence of communicating pseudocysts. Computed tomography was also obtained in our case and may be useful to determine the presenj::e of intrapancreatic or extrapancreatic f1.uid collections. In the present case, bronchoscopy was utilized in the initial evaluation of respiratory symptoms and provided direct visualization of the copious secretions coming from the left lower lobe. Deep lung secretions were collected and a pancreatic origin of the material was proven by determination of the isoenzyme pattern of the recovered amylase and by finding trypsinlike immunoreactivity in the fluid. Furthermore, successful diversion of pancreatic secretions was documented postoperatively by repeat bronchoscopy and the absence of pancreatic amylase or trypsinlike reactivity. Bronchoscopy with biochemical analysis of pulmonary secretions is a rapid way to diagnose a pancreaticobronchial fistula and provides useful information for optimizing respiratory support. Our case was treated by proximal drainage of a dilated pancreatic duct using a Roux-en-Y loop of jejunum. One other case used internal drainage of the fistula itself into the stomach with a successful outcome (4). The majority of cases reported used external drainage of the pancreas as the mainstay of

5 March 1986 PANcREATICOBRONCHIAL FISTULA 763 treatment (Table 2), although 1 case reported by Kaye (5) was treated successfully with tube thoracostomy alone. Distal pancreatectomy and thoracic drainage was added to abdominal drainage in several cases (Table 2). All of the reported cases were concluded successfully, with the exception of the case reported by Bell (7), in which the bronchial fistula was diagnosed after institution of mechanical ventilation for respiratory failure. Although adequate external drainage appears to be curative, direct internal drainage may considerably shorten the clinical course and alleviate complications. Our patient was discharged 3 wk after uncomplicated surgery and has remained well (follow-up of more than 6 mol. Pancrea.ticobronchial fistulas are unusual but should be included in a differential diagnosis of resistant pneumonias, particularly in the left lower lobe and when they complicate pancreatitis. The diagnosis is suggested by the presence of copious frothy sputum production and may be immediately diagnosed by bronchoscopy and determination of amylase activity in secretions. Endoscopic retrograde cholangiopancreatography is recommended to guide surgical drainage of the pancreas. Direct transabdominal external drainage appears to be a safe and successful means of treatment. If a dilated duct or a communicating pseudocyst is found by ERCP, internal drainage into the stomach, duodenum, or a defunctionalized jejunal limb is recom- mended. The outcome after adequate drainage should be highly satisfactory. References 1. Hunt RS. Pancreatico-bronchial fistula: a report of twb cases. Br J Surg 1954;41: Sease CI. Bronchial-pancreatic fistula: a case repbrt. Va Med Mon 1956;83: Ruffo A, Pironti L, Massa G. Pancreatico-bronchial fistula: description of a case. Minerva Med 1960;51: Dignan AP. Pancreatico-bronchial fistlila. Postgrad Med J 1965;41: Kaye MD. Pleuropulmohary complications of pancreatitis. Thorax 1968;23: Dubois J, Rousselle L, Piret R. Necrosing pancreatitis with pancreatico-bronchial fistula. Acta Chir Beig 1969;68: Bell JW. Pancteatico-bronchial fistula. Am Rev Resp Dis 1972;106:97~9. 8. Cox CL, Anderson IN, Guest JL. Bronchopancreatic fistula following traumatic rupture bf the diaphragm. JAMA HI77; 237: Grossman A, Jackson BT, Thompson RPH, Braimbridge MV. Pancreatico-broncho-pleural fistuia as a complication of acute pancreatitis. Br J Clin Pract 1978;32: Cameron JL. Chronic pancreatic ascites and pancreatic pleural effusions. Gastroenterology 1978;74: DaviS RE, Graham DY. Pancreatic ascites: the role of endoscopic pancreatography. Am J Dig Dis 1975;20: Filston HC, McLeod ME, Bolman RM, Jones RS. Improved management of pancreatic lesions in children aided by ERCP. J Pediatr Surg 1980;15:121-8.

Management of Pancreatic Fistulae

Management of Pancreatic Fistulae Jose Ramos University of the Witwatersrand Donald Gordon Medical Centre Fistula definition A Fistula is a permanent abnormal passageway between two organs (epithelial