Ann Thorac Surg 2002;73:1088-1091
© 2002 The Society of Thoracic Surgeons
Original article: general thoracic
Conservative management of thoracobiliary fistula
Bhugwan Singh, FCS (SA)*a,
Jaynathan Moodley, FCS (SA)a,
Mohamed H. Sheik-Gafoor, FCS (SA)a,
Naseem Dhooma, MBChB (Natal)a,
Anunathan Reddi, FCS (SA)b
a department of General Surgery, Faculty of Health Sciences, Nelson R. Mandela School of Medicine, University of Natal, Congella, South Africa
b department of Cardiothoracic Surgery, Faculty of Health Sciences, Nelson R. Mandela School of Medicine, University of Natal, Congella, South Africa
Accepted for publication December 18, 2001.
* Address reprint requests to Dr Singh, Department of Surgery, Faculty of Health Sciences, Nelson R. Mandela School of Medicine, University of Natal, Private Bag 7, Congella 4013, South Africa
e-mail: moodleyj6{at}nu.ac.za
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Abstract
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Background. Thoracobiliary fistulas are rare manifestations of biliary disruption. Given their rarity it is not surprising that there is little consensus on the optimal management of thoracobiliary fistulas.
Methods. Patients presenting with thoracobiliary fistulas over a 5-year period (1996 to 2001) were evaluated. Initial management was conservative with tube thoracostomy or drainage of sepsis when appropriate, or both; antibiotics and somatostatin were routinely administered. Endoscopic retrograde cholangiography was performed when symptoms persisted to delineate the thoracobiliary communication and undertake sphincteroplasty.
Results. Eight patients with a mean age of 31.9 years (range 15 to 42) were evaluated. Biliary effusion occurred in 3 patients after hepatic injury (n = 2) and percutaneous transhepatic cholangiography (n = 1). Bilioptysis occurred in 5 patients after hepatic abscess (n = 4) and hepatic injury (n = 1) The biliary effusion (n = 3) was successfully managed by endoscopic sphincterotomy in 2 patients; the third patient underwent urgent surgical biliary drainage. Bilioptysis (n = 5) was successfully managed in 3 patients; persistence of symptoms in 2 patients prompted surgical intervention.
Conclusions. Thoracobiliary fistulas may be successfully managed using a conservative approach. Surgery should be reserved for persistence of symptoms after exhaustion of this approach.
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Introduction
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Thoracobiliary fistulas (TBF), communications between the thorax and the biliary system, are rare. Graham [1] in 1897 originally described a TBF after thoracoabdominal trauma. TBF have since been reported to be congenital, to complicate inflammatory conditions (pyogenic [3, 4], amoebic [4, 5], echinococcal [4, 5, 6] or tuberculous [7]), neoplasms [8, 9] (first degree and second degree), and increasingly, to follow interventional procedures [4, 10, 11] on the biliary tract. TBF may communicate with either the pleural space (pleurobiliary) or the bronchus (bronchobiliary). Reports on this condition have largely been in the form of case reports. It is therefore not surprising that clear guidelines for the optimal treatment of TBF are lacking. In addressing this issue we report our experience in treating 8 patients with TBF.
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Patients and methods
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Patients presenting with TBF over a 5-year period (January 1996 to January 2001) to the surgical services at King Edward VIII Hospital, Durban, South Africa, were evaluated. The diagnosis of a TBF was confirmed by biochemical demonstration of elevated bilirubin levels in the pleural aspirate or sputum. Patients were begun on a low fat diet. The somatostatin analogue octreotide (100 µg 12 hourly) was administered subcutaneously. Broad-spectrum antibiotics were administered when there was clinical or radiologic evidence of sepsis. Metronidazole was administered to patients with amoebic liver abscess.
Abdominal ultrasonography and computed tomography (CT) was undertaken routinely to identify predisposing hepatic pathology. In cases of diagnostic ambiguity a di-isopropyl iminodiacetic acid scan (DISIDA) was performed.
Pleural biliary effusions were drained by tube thoracostomy. Abdominal collections, intrahepatic or subhepatic, were also drained percutaneously after ultrasonic localization. With the persistence of symptoms after medical treatment and drainage of sepsis, endoscopic retrograde cholangiography was performed to demonstrate biliary disruption, localize the site of the TBF, and exclude a distal biliary obstruction. Endoscopic sphincterotomy was routinely undertaken at the time of endoscopic retrograde cholangiography.
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Results
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Eight patients, 6 men and 2 women, were diagnosed with a thoracobiliary fistula. Their mean age was 31.9 years (range 15 to 42). Five patients presented with bilioptysis ranging from bile stained sputum to copious amounts of frank bile. Three patients presented with progressive dyspnea due to massive biliary effusions.
The predisposing pathology is outlined in Table 1.
The 3 patients who sustained thoracoabdominal trauma (patients 5, 6, and 7) had undergone laparotomy initially; the hepatic injuries were managed by hepatorrhaphy and drainage. Patient 8 had undergone percutaneous transhepatic cholangiography for the evaluation of obstructed jaundice that developed after biliary surgery.
Abdominal sonography and CT confirmed an intrahepatic abscess in 4 patients. In patient 2, CT demonstrated air lucencies not typical for uninfected amoebic liver abscess, thereby suggesting a thoracic communication (Fig 1).
Percutaneous drainage was undertaken in these patients with symptomatic improvement of the abdominal signs but not the bilioptysis.
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Fig 1. Computed tomography scan demonstrating an amoebic liver abscess containing air lucencies (arrow) suggesting thoracic communication.
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A DISIDA scan was undertaken in patient 6; although the pleural communication was demonstrated, this did not contribute to the patient’s further management (Fig 2).
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Fig 2. Di-isopropyl iminodiacetic acid scan demonstrating isotope in the right pleural space (arrow) suggesting thoracobiliary fistula.
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Endoscopic retrograde cholangiogram and sphincterotomy was undertaken in 7 patients when the symptoms persisted. Endoscopic retrograde cholangiogram was performed, on the mean at day 12 (range 9 to 16) after the diagnosis of a thoracobiliary fistula. In 2 patients, both with bilioptysis, a fistulous tract extending to the basal segments of the right lung was demonstrated (Fig 3); in the remaining 5 patients no fistulous tract could be demonstrated.
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Fig 3. Endoscopic retrograde cholangiography demonstrating contrast outlining an abscess cavity (black arrow); note thoracobiliary communication (white arrow).
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There was a dramatic improvement in symptoms (within 72 hours) after sphincterotomy in 5 patients (patients 2, 3, 4, 6, and 7). The 2 patients with persistent symptoms underwent laparotomy with drainage of hepatic collections and repair of the diaphragm. One patient required a relaparotomy to repair a persistent fistula and diaphragmatic defect that necessitated the use of a mesh repair. Recovery was uneventful in both patients.
Patient 8 did not undergo endoscopic retrograde cholangiography. In this patient a biliary stricture was suspected and urgent surgical intervention for biliary sepsis was necessary. A choledochojejunostomy was fashioned. Resolution of the biliary pleural effusion occurred rapidly thereafter.
In the 5 patients managed successfully by endoscopic sphincterotomy the mean hospital stay was 18.6 days (range 13 to 22); in the 3 patients managed surgically the mean hospital stay was 33 days (range 25 to 42). No recurrence of symptoms was reported at the 3- and 6-month follow-up. The mean follow-up was 26 months (range 2 to 48).
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Comment
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Early diagnosis of a TBF is crucial in the management of this condition. A delayed diagnosis allows complications to develop that may warrant extensive surgery. Bile has been shown to have a corrosive effect upon the lung and pleural space [12, 13]. A high index of suspicion in the appropriate clinical situation is therefore mandatory. The presence of bile on thoracentesis of a pleural effusion and bilioptysis are pathognomonic for TBF. Bilioptysis may range in presentation from bile-stained sputum to the expectoration of large volumes of bile occasionally approaching a liter [8]. Pleurobiliary fistula may predispose to a loculated bilious empyema; the consequent development of pleural adhesions may entrap the lung, compromising lung function [4]. Bronchobiliary fistula may predispose to a necrotizing bronchitis or bronchopneumonia; rarely a chronic indirect pneumonitis may develop [8].
Abdominal sonography is the first investigation in our management policy (Fig 4).
Abscesses and biliary collection are readily identified and may be drained percutaneously. However, neither CT nor ultrasonography is capable of delineating the precise location of the thoracobiliary fistula that is necessary should surgery be planned. Similarly, while radionucleide scanning has been shown to demonstrate the presence of TBF it fails to provide information on its precise anatomic location. Endoscopic cholangiography may demonstrate the fistulous tract and identify distal biliary obstruction, crucial for the persistence of TBF. Furthermore, endoscopic sphincterotomy may be undertaken during this study. The authors recommend endoscopic sphincterotomy when there is persistence of symptoms within 72 to 96 hours of the placement of tube thoracostomy or percutaneous drainage of sepsis. Endoscopic sphincterotomy alone has been applied successfully to treat bronchobiliary fistula complicating gallstones [25], biliary fistula complicating hepatic trauma [26, 27], and biliary surgical procedures [28]. Geenen and colleagues [29] noted the normal common bile duct pressure to be 10 ± 2 mm Hg and the basal sphincter of Oddi pressure to average 18 ± 3 mm Hg; superimposed on basal sphincter of Oddi pressure are prominent phasic contractions with an amplitude up to 124 ± 16 mm Hg with a rate of 4 ± 1 contractions per minute. The common bile duct to duodenum gradient before sphincterotomy was noted to be 10 ± 1 mm Hg [21]. Given these pressure gradients, in an intact ductal system bile flows along an open fistula tract following the path of least resistance. After sphincterotomy the mean basal sphincter of Oddi pressures decreases to 1 ± 1 mm Hg [29] favoring the prograde flow of bile into the duodenum rather than into the fistula tract. This provides the rationale for endoscopic sphincterotomy that was successful in 5 of the 7 patients in whom the procedure was undertaken, the symptoms resolving within 48 hours. Pharmacologic agents that lower the sphincter of Oddi pressure and thereby facilitate biliary decompression have been suggested [21]; their effectiveness and role in managing thoracobiliary fistula is presently unclear.
Definitive surgery for a bronchobiliary fistula entails excision of the fistula tract and lung tissue destroyed by the necrotizing bronchopneumonia; this may entail a wedge resection, segmentectomy, or lobectomy. Early surgical intervention has been advocated to circumvent the respiratory compromise to which these fistulas are liable to predispose [14, 15]. In contrast pleurobiliary fistula is generally a less sinister condition; a conservative approach with tube thoracostomy drainage and appropriate antibiotics has been reported to be successful in as many as 60% of posttraumatic pleurobiliary fistula cases [16]. While resolution with a conservative approach may be prolonged, this may be balanced against the prospect of a formidable surgical procedure. There is consensus that during the period of diagnostic workup conservative therapy does not impart additional morbidity should subsequent surgery become necessary [17]. In addition to tube thoracostomy, percutaneous drainage of subphrenic collections, antibiotics, and somatostatin have been successfully used in the management of biliary fistula [18, 19].
The importance and benefits of biliary decompression in TBF are further substantiated by the reports on temporary transampullary stenting that have shown it to significantly decrease morbidity and mortality when compared with surgical intervention [19, 20]. Stents are removed after healing of the fistula, usually within 6 weeks, which corresponds with a patency period of small calibre stents [21]. The effectiveness of endoscopic therapy has been underscored by the successful treatment of 11 patients with bronchobiliary fistulas using endoscopically placed nasobiliary drains [6]. The application of long stents to bridge intrahepatic biliary defects is controversial [22]; however, there are some who advocate placement of biliary endoprosthesis over biliary defects to facilitate fistula closure [19, 23, 24].
In current surgical practice the wide availability of interventional endoscopy has enhanced the conservative approach to managing TBF. The favorable impact of endoscopic sphincterotomy has been demonstrated in this series. In addition to its early diagnosis, it is suggested that TBF, after the failure of medical treatment, be managed by endoscopic sphincterotomy before recourse to surgery. [2]
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Abstract
Introduction
