- Case report
- Open Access
Egyptian Liver Journal volume 9, Article number: 4 (2019)
Biliptysis means coughing of bile which is a presenting symptom of a rare condition called bronchobiliary fistula (BBF). BBF is a connection between the biliary tract and bronchial tree. BBF mostly occurs secondary to malignancy, liver abscess, and trauma. Surgical approach in BBF management was the main management strategy, then endoscopic approach.
We managed our first encountered case of biliptysis endoscopically by endoscopic retrograde cholangiopancreatography (ERCP).
ERCP management seems to be effective in management of biliptysis.
Bronchobiliary fistulas are rare. In most cases, they are caused by neoplasms and hepatic or subphrenic abscesses, resulting from different conditions or trauma . It is usually diagnosed by clinical history (coughing of bile) and imaging (CT/MRI) [2, 3]. Treatment is usually surgery or endoscopic or transhepatic embolization [4, 5]. We used an ERCP endoscope and placing of stent for appropriate closure of the fistula.
A 26-year-old male working as a cook in a restaurant presented with a history of fever and abdominal pain for 2 weeks and received therapy in the form of empiric antibiotics and antipyretics with no improvement. Later, the patient started to complain from coughing of dark yellowish sputum (biliptysis). A CT scan on the abdomen and lower chest was ordered and revealed large subphrenic abscess (Fig. 1). Then, the patient was referred to our hospital (National Liver Institute (NLI), Shebin Alkawm, Egypt) and was admitted to the surgery department. CT of the abdomen was repeated and confirmed the subphrenic abscess connected to the lower lung zone abscess with right hepatic lobe abscess. Aspiration of fluid sample from the abscess for bacteriological evaluation revealed pure pus with negative culture for any organism (may be related to how much antibiotics the patient had received). Liver profile was normal which is a rare finding in such hepatic lesions. Kidney function was also normal. He had negative virology markers (HBV, HCV, and HIV).
The patient was referred to our department (hepatology and gastroenterology) to give him a trial of endoscopic management. ERCP was done, and the fluoroscopic image revealed pooling of contrast in the right hepatic lobe (abscess) with contrast seen tracking upward to the lower lung (Fig. 2 is a picture of bronchobiliary fistula). Cannulating an intrahepatic small biliary duct by guidewire to bypass the site of fistula and drain the hepatic abscess was successful, and this was followed by inserting a 10–15-cm plastic stent. Improvement of biliptysis was achieved which is associated with improvement of fever. The patient was discharged from our institute 5 days after endoscopy and was followed up at the surgery department weekly for 3 months. His symptoms totally improved, and follow-up CT showed resolution of the abscess.
BBF, despite being rare, is known to be caused by different causes: congenital, malignancies, abscesses, traumatic, or iatrogenic [4, 6]. Diagnosis is usually made clinically for biliptysis and it is sometimes inappropriately diagnosed as pneumonitis or chronic cough with greenish sputum  .
Somatostatin and its analogues were tried for treating BBF, by reducing digestive tract secretions . Patients should be advised to take orthostatic position and avoid supine position to decrease the volume of coughed bile and accelerate fistula healing. Also, supporting therapy should be administered with appropriate prophylactic attention to electrolyte disturbance .
Definitive treatment for BBF has not yet been established. Surgical or non-surgical interventional procedures, such as endoscopic retrograde cholangiopancreatography (ERCP) or percutaneous transhepatic drainage (PTD), are frequently used as direct photographic evidence and management .. Transhepatic embolization, bronchoscopic injection of n-butyl cyanoacrylate, or histoacryl embolization has been tried [4, 7]. A systematic review done on 68 cases had reported that interventional procedures were slightly more effective than surgical procedure (97% vs. 85%) .
In the case of BBF due to abscess, we think two combined approaches could be attempted. The first approach is abscess management by antibiotics and drainage, and the second one is biliary drainage either by ERCP or PTD. Shrinking the abscess cavity closes the fistula tract, and biliary drainage prevents the recurrence of the fistula.
ERCP stenting (endoscopic approach) is easy and is a possible management for cases of biliptysis.
Availability of data and materials
The data and material are available.
Endoscopic retrograde cholangiopancreatography
Percutaneous transhepatic drainage
SS AL-M, HH AL-J (1999) Chronic cough due to bronchobiliary fistula. Respiration 66(5):473–476. https://doi.org/10.1159/000029415
Liao GQ, Wang H, Zhu GY, Zhu K Bin, Lv FX, Tai S. Management of acquired bronchobiliary fistula: a systematic literature review of 68 cases published in 30 years. World J Gastroenterol 2011;17(33):3842–3849. doi:https://doi.org/10.3748/wjg.v17.i33.3842
Ragozzino A, De Rosa R, Galdiero R, Maio A, Manes G (2005) Bronchobiliary fistula evaluated with magnetic resonance imaging. Acta Radiol 46(5):452–454 http://www.ncbi.nlm.nih.gov/pubmed/16224917
Mehrzad H, Aziz A, Mangat K (2012) Transhepatic embolisation of a traumatic broncho-biliary fistula: a novel approach. Case Reports 2012(nov27 1):bcr2012006702–bcr2012006702. https://doi.org/10.1136/bcr-2012-006702
Yilmaz U, Sahin B, Hilmioglu F, Tezel A, Boyacioglu S, Cumhur T (1996) Endoscopic treatment of bronchobiliary fistula: report on 11 cases. Hepatogastroenterology 43(7):293–300 http://www.ncbi.nlm.nih.gov/pubmed/8682482
Fröbe M, Kullmann F, Schölmerich J, Böhme T, Müller-Ladner U (2004) Bronchobiliäre Fistel bei kombiniertem Lungen-Leber-Abszess. Med Klin 99(7):391–395. https://doi.org/10.1007/s00063-004-1057-4
Shim JR, Han S-S, Park HM, Lee EC, Park S-J, Park J-W (2018) Two cases of bronchobiliary fistula: case report. Ann Hepato-Biliary-Pancreatic Surg 22(2):169. https://doi.org/10.14701/ahbps.2018.22.2.169
Ong M, Moozar K, Cohen LB (2004) Octreotide in bronchobiliary fistula management. Ann Thorac Surg 78(4):1512–1513. https://doi.org/10.1016/j.athoracsur.2003.10.137
Karabulut N, Çakmak V, Kiter G (2010) Confident diagnosis of bronchobiliary fistula using contrast-enhanced magnetic resonance cholangiography. Korean J Radiol 11(4):493. https://doi.org/10.3348/kjr.2010.11.4.493
Thanks for my dear professor Dr. Gamal Badra Who gave me his experience in Endoscopy.
Ethics approval and consent to participate
This study was approved by our institutional review board (IRB) (IRB00003413).
Consent for publication
Written informed consent was obtained from the patient for publication of this case report and accompanying images.
The author declares that he has no competing interests.
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
About this article
Cite this article
Elbahr, O. Biliptysis. Egypt Liver Journal 9, 4 (2019). https://doi.org/10.1186/s43066-019-0005-8