A 48-year-old white woman, who worked in agriculture, was admitted
with a 4-month history of cough productive of bile-like yellow phlegm. She had
been hospitalized several times previously for recurrent right-sided
pneumonias. The patient had undergone surgical resection of echinococcal cysts
within liver segments V, VI, and VIII, cholecystectomy, and reconstruction of
the right hepatic duct damaged by parasitic cyst infiltration 6 years
previously ([Fig. 1]).
Fig. 1 Abdominal computed
tomography (CT) image of an echinococcal cyst in the right lobe of the
liver.
External drains had been inserted into both hepatic ducts through
the common bile duct under radiological guidance ([Fig. 2]).
Fig. 2 Postoperative
cholangiography showing leakage of bile from the region of the right hepatic
duct.
At subsequent outpatient visits, leakage of bile from the drains had
persisted, and 3 months later, a plastic stent had been inserted into each
hepatic duct during endoscopic retrograde cholangiopancreatography (ERCP). The
patient had been advised to attend again for monitoring within
3 – 4 months with the possibility of replacement stents
being required. However, she had refused to continue undergoing endoscopic
procedures, and the stents remained in place for the next 6 years.
At the time of readmission, abdominal and
chest computed tomography (CT) showed inflammatory lesions and a fluid
collection in the subdiaphragmatic region; it was impossible to pinpoint the
exact location of the fistula at bronchoscopy. A subsequent CT and ERCP
demonstrated the fistula between the biliary system and the lower lobe of the
right lung ([Figs. 3]
[Figs. 4]
[Figs. 5]).
Fig. 3 Abdominal and chest
computed tomography (CT) image showing the presence of the fistula between the
biliary system and the lower lobe of the right lung (arrow a) and the
previously placed plastic biliary stents (arrow b).
Fig. 4 Both stents are seen
protruding from the major duodenal papilla during endoscopic retrograde
cholangiopancreatography (ERCP).
Fig. 5 The presence of the
fistula between the biliary system and the lower lobe of the right lung (red
arrows) is shown during endoscopic retrograde cholangiopancreatography
(ERCP).
After endoscopic removal of the occluded plastic biliary stents, the
patient was treated with antibiotics, and an improvement in her general
condition and regression of symptoms were observed. A follow-up thoracic CT
showed no inflammatory lesion in the lung tissue ([Fig. 6]).
Fig. 6 Thoracic computed
tomography (CT) image after antibiotic therapy and endoscopic removal of the
plastic biliary stents showing no signs of inflammatory lesions in the
lungs.
The patient was discharged home after 8 days in good general
condition. There were no recurrent symptoms at follow-up 24 months later.
A bronchobiliary fistula (BBF), a communication of the biliary
system with the bronchial tree, is an exceptionally rare condition with
underlying factors that include hydatid disease, hepatobiliary surgery, hepatic
trauma, and congenital malformation [1]
[2]
[3]
[4]. The
mechanism of transdiaphragmatic extension in BBF remains controversial. BBF due
to the migration of short-term intrahepatic biliary stents has been previously
reported [5]. To our knowledge, BBF caused by long-term
extrahepatic biliary stenting has not previously been described, but has
clearly been shown by our case, which also demonstrates that in particular
cases, the condition can be successfully managed with a conservative endoscopic
approach.
Endoscopy_UCTN_Code_CPL_1AK_2AI
