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Bronchobiliary fistula: a case report.

CASE REPORT

A 52-year-old woman presented to the hospital with a six week history of productive cough, wheezing, and shortness of breath. She reported no fever or chill. She had a history of reactive airway disease, gastroesophageal reflux disease, and a 30-pack-year smoking history. The patient had stopped smoking one and a half years ago. She had been treated for stage III colon cancer six years ago with colon resection and chemotherapy consisting of Folfox (folinic acid, fluorouracil, and oxaliplatin) and Avastin (bevacizumab). Five years after treatment, she underwent right hepatic lobectomy for an isolated metastasis. Her hepatic resection was complicated by postoperative subdiaphragmatic abscess. One year following right hepatic resection, she complained of unrelenting cough with yellowish-green sputum, chest pain, and hoarseness. She was treated for presumed pneumonia with multiple intravenous and oral antibiotics, bronchodilators, and steroids without resolution of her symptoms.

During this admission, the patient underwent sputum assay which was positive for bilirubin; and the sputum grew Klebsiella pneumoniae. The patient also had leukopenia (2,700/[mm.sup.3]) and mild anemia. Her hepatic function panel was within normal limits. The patient was evaluated for a bronchobiliary fistula with computed tomography (CT) scan of the chest and abdomen (Figures 1, 2), and a dimethyl iminodiacetic acid (HIDA) scan (Figure 3).

The patient was diagnosed with bronchobiliary fistula, and she went to operation. Her preoperative pulmonary function tests were in the 90th percentile. She underwent a right thoracotomy, dissection of right lower lobe off of an inflamed area of diaphragm, and right lower lobectomy. No bile leak was seen inside the pleural space. Intraoperative frozen section showed no malignancy at the suspected fistula site. The abnormal diaphragmatic surface was sealed with bioglue, and a vascularized parietal pleural flap was created and tacked over the diaphragm with permanent suture (Figure 4). A chest tube and Blake drain were placed, and the right lower lobe was sent for pathoanatomical examination.

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The lobectomy specimen demonstrated chronic inflammation and a fistulous tract containing blood and bile. The patient also had to two foci of metastatic adenocarcinoma. The patient had an uneventful postoperative course with resolution of her cough. She was discharged from the hospital on postoperative day eight. She is now five months postoperative without recurrence of symptoms.

DISCUSSION

The common causes of bronchobiliary fistula are rupture of a hydatid cyst or erosion of an abscess through the diaphragm. (3) Often an obstruction of the biliary tree is present. The pressure gradient between the bile ducts and bronchi keeps the fistulous tract open. These patients often present with cough and chest pain secondary to the inflammatory reaction caused by biliptysis. The sputum can be secondarily infected, and the bile-tinged sputum often is mistaken for more common reactive sputum. Due to the rarity of this condition, there have been many approaches to treatment.

Patients with an active abscess should undergo surgical intervention, but in the majority of cases, these fistulas are chronic, and can be treated with a less invasive approach. Current literature has recommended endoscopic retrograde cholangiopancreatography (ERCP) and stent placement for diagnosis and treatment with surgical intervention reserved for treatment failures. The stenting of the Sphincter of Oddi may decrease biliary pressure and promote tract healing by decreasing the rate of bile flow to the lungs. Normal common bile duct pressure is 10 [+ or -] 2 mm Hg, and drops to 1 [+ or -] 1 mm Hg after sphincterotomy. (4) Small studies have recommended combining octreotide acetate and a low fat diet with stenting to decrease hepatic bile production. (5) Other studies have used fluoroscopy to place coils, glue, or fibrin sealant into the fistula tract. (6,7,8) Biliary stents will stay in place for approximately six weeks to allow for healing, and then will need endoscopic retrieval due to the high occlusion rate of these small lumen stents. In our patient, we felt the cause of the fistula could be recurrence of her cancer and would necessitate surgical intervention. This fistula was not caused by a malignancy, but metastatic lesions were found in the lobectomy specimen.

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Physicians should have a high index of suspicion of bronchobiliary fistula in patients presenting after hepatic intervention with cough and yellow-green sputum that is not ameliorated with antibiotics. The workup for bronchobiliary fistula should start with a sputum assay for bile, followed by an imaging modality such as CT, ERCP, or HIDA. The physician should then proceed with a plan of ERCP/sphincterotomy or surgical intervention. It is likely that bronchobiliary fistula rates will increase as the number of hepatic interventions continues to rise.

REFERENCES

(1.) Peacock TB. Case in which hydatid cyst of the liver, communicating with the lung. Read before the Royal Medico-Chirurgical society, March 1850.

(2.) Gugenheim J, Ciardullo M, Traynor O, et al. Bronchobiliary fistulas in adults. Ann Surg 1988;207:90-94.

(3.) Eryigit H, Oztas S, Urek S, et al. Management of acquired bronchobiliary fistula: 3 case reports and a literature review. J Cardiothorac Surg 2007;2:52.

(4.) Singh B, Moodley J, Sheik-Gafoor MH, et al. Conservative management of thoracobiliary fistula. Ann Thorac Surg 2002;73:1088-1091.

(5.) Ong M, Moozar K, Cohen LB. Octreotide in bronchobiliary fistula management. Ann Thorac Surg 2004;78:1512-1513.

(6.) Chua HK, Allen MS, Deschamps C, et al. Bronchobiliary fistula: principles of management. Ann Thorac Surg 2000;70:1392-1394.

(7.) Vimalraj V, Jeswanth S, Selvakumar E, et al. A case of recurrent biliptysis. J Thorac Cardiovasc Surg 2007;133:1662-1663.

(8.) Goldman SY, Greben CR, Setton A, et al. Bronchobiliary fistula successfully treated with n-butyl cyanoacrylate via bronchial approach. J Vasc Interv Radiol 2007;18:151-155.

Ian A. Hodgdon, MD; and R. Scott Thurston, MD

Dr. Hodgdon and Thurston are affiliated with the Louisiana State University Health Sciences Center, Department of Surgery, New Orleans, and Our Lady of the Lake Regional Medical Center, Department of Cardiothoracic Surgical, Baton Rouge.
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Author:Hodgdon, Ian A.; Thurston, R. Scott
Publication:The Journal of the Louisiana State Medical Society
Article Type:Case study
Geographic Code:1U7LA
Date:May 1, 2011
Words:988
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