Late onset of biliopleural fistula following percutaneous transhepatic biliary drainage: a case report
Late onset of biliopleural fistula following percutaneous transhepatic biliary drainage: a case report
Edward Yi-Yung Yu 1 2
Fei-Shih Yang 2
Yu-Jen Chiu 0
Fuu-Jen Tsai 4 5 6
Chi-Cheng Lu 3
Jai-Sing Yang 7
0 Division of Reconstructive and Plastic Surgery, Department of Surgery, Taipei Veterans General Hospital , Taipei 112 , Taiwan
1 Department of Radiology, Taitung MacKay Memorial Hospital , Taitung 950 , Taiwan
2 Department of Radiology, MacKay Memorial Hospital , Taipei 104 , Taiwan
3 Department of Pharmacy, Buddhist Tzu Chi General Hospital , Hualien 970 , Taiwan
4 Department of Medical Genetics, China Medical University Hospital , Taichung 404 , Taiwan
5 School of Chinese Medicine, China Medical University , Taichung 404 , Taiwan
6 Genetics Center, Department of Medical Research, China Medical University Hospital , Taichung 404 , Taiwan
7 Department of Medical Research, China Medical University Hospital, China Medical University , Taichung 404 , Taiwan
Biliopleural fistula (BF) and formation of biliopleural effusion is a rare complication following percutaneous transhepatic biliary drainage (PTBD). It occurs when the pleura is traversed by the catheter before entering the bile duct. Biliopleural fistula should be suspected when right side pleural effusion develops following the PTBD procedure. The diagnosis of biliopleural fistula is made when greenish pleural fluid with high concentration of bilirubin is aspirated. Here we present a case where a patient develops a biliopleural fistula following PTBD due to obstructive jaundice caused by neuroendocrine tumor of pancreas. Biliopleural fistula was disclosed after a scheduled catheter replacement procedure. Treatments of biliopleural fistula include thoracentesis with drainage tube installation into pleural space. In addition, a drainage tube was installed through percutaneous transhepatic gallbladder drainage (PTGBD) to reduce the bile induced pressure. Surgical repair of fistula was performed after the conservative treatment was unsuccessful. The patient expired 5 days after surgery due to respiratory failure.
Biliopleural fistula (BF); Percutaneous transhepatic biliary drainage (PTBD); Neuroendocrine tumor; Jaundice
Biliopleural fistula (BF) and the formation of biliopleural effusion
is a rare complication of percutaneous transhepatic biliary drainage
]. It occurs when the pleural cavity is traversed during
the procedure to gain access to the biliary tract. The likelihood of
fistula formation increases with the duration of catheter placement [
Here we present a case of biliopleural fistula which was
disclosed 98 days after the initial PTBD procedure. Our objective of
this case is to increase the awareness of interventional radiologists
of this rare complication that may lead to significant morbidity
and even mortality.
2. Case Presentation
A 53-year-old woman with a clinical history of neuroendocrine
tumors in pancreas head with hepatic metastasis was admitted to
our center due to recent onset of jaundice. Abdominal computed
tomography (CT) revealed pancreas head tumor, multiple hepatic
metastasis and dilatation of intra hepatic bile duct (IHD) and
common bile duct (CBD).
Percutaneous transhepatic biliary drainage (PTBD) was
performed as a palliative treatment to reduce bile induced jaundice.
Right side transhepatic approach was performed with a needle
inserted through midaxillary line between 9th and 10th rib. An
8 French (FR) multiple side hole, a pig-tailed was inserted into
CBD for continuous drainage. Abdominal CT was arranged the
next day, and there was no evidence of procedure-related
complications (Fig. 1). The positioning of the catheter was followed up
regularly with chest X-ray film and plain abdominal film without
evidence of catheter migration. A follow up PTBD was
performed 45 days after the initial PTBD, and without evidence of
catheter migration (Fig. 2). Scheduled catheter replacement was
arranged 98 days after the initial PTBD procedure. A new
catheter the same caliber was inserted into the CBD without technical
difficulty (Fig. 3).
This patient developed high fever 8 days after returning to
the ward. Chest X-ray film revealed right side pleural effusion
(Fig. 4). Thoracentesis was performed and greenish fluid was
collected. A drainage tube was installed into right pleural space
for continuous drainage. Biochemical report of the greenish fluid
revealed a high concentration bilirubin (23.7 mg/dl). Bacteria
culture revealed Enterococcus and Candida albicans.
Appropriate antibiotics were given accordingly.
Follow-up of PTBD was performed the next day, and BF
with back flow of contrast media (CM) into right pleural space
was disclosed (Fig. 5). Additional installation of drainage tube
via percutaneous transhepatic gallbladder drainage (PTGBD)
was performed 3 days later due to no sign of decreasing drainage
amount of right pleural effusion. Surgical repair of the fistula was
arranged 4 days later due to persistent right pleural effusion. The
patient expired 5 days after surgery due to respiratory failure.
Percutaneous transhepatic biliary drainage is often used in
treatment of obstructive biliary disease to relieve symptoms prior to
surgery or palliatively in patients who are poor candidates for
]. Biliopleural fistula is a rare complication
following this procedure, which may be due to the passage of catheter
through the pleural cavity before crossing the diaphragm and
into the bile duct when a transhepatic approach is used [
Elevated pressure gradient in the biliary tract could drive the bile
leak back into the pleural cavity. The likelihood of fistula
formation between the biliary tract and pleural cavity increases with the
duration of catheter in place and is the primary factor leading to
fistula formation . Studies have shown that fistula formation
occurs within 3 weeks of catheter placement and biliopleural
effusion may develop when the catheter remained in bile duct for
more than 4 weeks [
]. One study revealed common features that
lead to the development of the BF: (i) Complete biliary
obstruction was present; (ii) Catheter placement was between the 9th and
10th ribs in the midaxillary line; (iii) prolonged drainage (7 days
to 2 months) preceded fistula formation [
]. These features are
also seen in our patient. Another study suspected the path created
by the large drainage tubes served as an ideal passage through
which bile could leak back into the pleural cavity in the presence
of persistent biliary tract obstruction [
]. Bilious fluid collections
can be present anywhere along the path of the PTBD catheter
from the biliary tree to the pleural space [
We suspect the cause for the late onset of BF and
biliopleural effusion in this patient: (A) high efficiency of the catheter in
draining the bile juice from CBD; (B) the residual blood clot and
the infected bile juice with much debris accumulated at the path
of the catheter from the initial PTBD procedure, which acts as a
sealant forming between the catheter and the adjacent liver
tissue to prevent the back flow of bile juice from the drainage tract
into the pleural space. Removal of the original catheter causes
the sealant to dislodge from the drainage tract; this creates a tiny
space between the new catheter and the adjacent liver tissue,
leading to the back flow of bile juice from CBD, around the catheter,
and into pleural space.
A high percentage of patients in other series and in our patient
developed empyema. The reasons for infectious complications
are multiple. The direct tract from the skin to the pleura without
true pleural drainage could predispose to pleural seeding with
bacterial pathogens [
]. The incidence of cholangitis in patients
with completely obstructed and dilated biliary tracts is high
(> 80%) without clinical symptoms [
]. The patient should be
evaluated for empyema if a BF develops.
Early diagnosis of BF can reduce complications requiring
]. The diagnosis of BF should be suspected when a
patient with a PTBD catheter develops a right pleural effusion.
Ultrasound, CT and radionuclide scan can identify bile collection,
but they cannot determine fistula location [
]. Confirmation is
obtained on thoracentesis when bilious green fluid aspirated that
has a pleural fluid total bilirubin to serum total bilirubin ratio >1.0
]. Follow-up cholangiogram may sometimes reveal the
fistulous tract when the CM regurgitates through the side hole of the
catheter and into the pleural cavity.
The treatment for BF is surgical; although few patients
tolerate surgical intervention, it can also be treated with conservative
]. Bile drainage from the pleural cavity can be
done conservatively with a drainage tube. Early institution of
another form of biliary drainage appears to be the single most
important factor in the successful management of BF . One
study with surgical experience with traumatic BF revealed that
chest drainage ceases or dramatically decreases once an
alternative route of biliary decompression is available [
]. Surgery is
required if the fistula is large or diseased tissue must be debrided
]. The application of antibiotics as an adjunct to bile drainage
is critical because: (i) bacteria can enter along the catheter path; (ii)
bile is often infected; (iii) bile stasis encourages bacteria growth
]. Because the likelihood of fistula formation is related to the
length of time the catheter is in place, prevention relies on
decreasing the duration of the time the catheter is in place.
Biliopleural fistula and biliopleural effusion is a rare but serious
complication of PTBD. It should be suspected in patient who
develops right side pleural effusion after PTBD. This complication
should be treated conservatively, and surgical treatment should be
performed if the conservative treatment has failed.
Declaration of Conflicting Interests
The author(s) declared no potential conflicts of interest with
respect to the research, authorship, and/or publication of this article.
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