Brief Report

The American Surgeon

Surgical Management of
2022, Vol. 88(6) 1 343­–1345
© The Author(s) 2020
Article reuse guidelines:
Bronchobiliary Fistula After sagepub.com/journals-permissions
DOI: 10.1177/0003134820945263
Thoracoabdominal Trauma journals.sagepub.com/home/asu

Julia M. Coughlin, MD1,2, Steven Bonomo, MD1,2, Edie Y. Chan, MD1,2,

Jafar Hasan, MD3, Mark A. Grevious, MD3, and Nicole Geissen, DO4

Abstract

Bronchobiliary fistulas are exceedingly rare pathological connections between the biliary and the bronchial systems,
which result from hepatobiliary neoplasms, abscesses, or thoracoabdominal trauma. Prompt recognition, diagnosis,
and intervention is essential in order to prevent the high morbidity and mortality associated with this disease process.
Multiple management strategies have been described in the literature; however, the optimal course has not been well
defined. We present a case of a 31-year-old male who developed a bronchobiliary fistula 1 month after thoracoabdom-
inal trauma. After conservative management with biliary stenting failed, he successfully underwent latissimus sparing
right posterolateral thoracotomy, complete fistulectomy, right lower lobe wedge resection, and diaphragmatic recon-
struction with subsequent resolution of his symptoms.

Keywords
bronchobiliary fistula, penetrating trauma, hepatobiliary

A 31-year-old Hispanic male presented after he sustained a
gunshot to the thoracoabdomen. He underwent immediate
surgical exploration and was found to have an injury to the
posterior dome of the liver between segments 7 and 8, as
well as between segments 5 and 6. Additionally, he sus-
tained injury to the right hemi-diaphragm and right colon at
the hepatic flexure. He underwent primary repair of the dia-
phragmatic injury, right hemicolectomy with primary anas-
tomosis, small bowel resection, and repair of the right liver
laceration. His postoperative course was complicated by
evisceration, which required multiple returns to the operat-
ing room for abdominal washout and temporary closure.
Approximately 28 days after his initial operation, he devel-
oped low grade fevers, persistent cough, and biliptysis. A
computed tomography (CT) scan demonstrated a right
pleural effusion, a right sub-phrenic fluid collection, and
possible communication with the right biliary system.
During drain placement by interventional radiology (IR), he
was noted to have a communication between the two fluid
collections. He underwent endoscopic retrograde cholan-
giopancreatography (ERCP), which demonstrated a bron-
chobiliary fistula (Figure 1). Biliary stent placement
temporarily relieved his biliptysis. Despite upsizing of the
stent, he continued to have a persistent productive cough
with large volume biliptysis. He was transferred to our insti-
tution for definitive surgical intervention.
Prior to his operation, a multidisciplinary surgical con-
ference was held with several surgical specialists in order
to discuss the optimal surgical approach. It was deter-
mined that a latissimus sparing right posterolateral thora-
cotomy would be performed. Immediately upon entrance
into the thoracic cavity, he was noted to have a thick rind
encasing the right lower and middle lobes. Decortication
was performed and the lower lobe was found to be densely
adherent to the diaphragm. The fistulous tract was
1
Department of General Surgery, Rush University Medical Center,
Chicago, IL, USA
2
Department of General Surgery; John H. Stroger, Jr. Hospital of
Cook County, Chicago, IL, USA
3
Department of Plastic Surgery; John H. Stroger, Jr. Hospital of Cook
County, Chicago, IL, USA
4
Department of Thoracic Surgery; John H. Stroger, Jr. Hospital of
Cook County, Chicago, IL, USA
Corresponding Author:
Julia M. Coughlin, MD, Department of General Surgery, Rush
University Medical Center, Chicago, IL 60647, USA.
Email: Julia_m_Coughlin@rush.edu
2
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Figure 1. Endoscopic retrograde cholangiopancreatography.
Visualization of contrast within the biliary tree communicating
with the right lower lobe.
identified, an elliptical incision was made on the dia-
phragm, and the fistula was followed to segment 7 of the
liver (Figure 2A). With a probe, the tract was found to
connect to the right common hepatic duct. A complete fis-
tulectomy was performed, which included the portions of
involved liver and diaphragm. A drain was placed in the
liver defect and multiple additional perihepatic drains
were secured. A wedge resection of the right lower lobe
was also performed. The entire specimen was removed en
bloc (Figure 2). The diaphragmatic defect measured
approximately 5 cm in diameter. The distal portion of the
latissimus dorsi flap was utilized to obliterate the dead
space from the liver resection while the proximal portion
was used to reconstruct the diaphragmatic defect.
Postoperatively, the patient’s pulmonary symptoms
resolved immediately. Drains were sequentially removed
at postoperative clinic visits over a course of six months.
He has since been asymptomatic and expected to make a
full recovery.
Acquired bronchobiliary fistula is a rare complication
that can develop secondary to hepatobiliary surgery,
hepatic infection, neoplasm, biliary obstruction, or thora-
coabdominal trauma.1-4 Penetrating thoracoabdominal
trauma creates the ideal environment for the development
of bronchobiliary fistulas due to the direct connection
between the commonly injured organs, which include the
lower lobe of the right lung, the diaphragm, and the liver.
In some cases, right-sided diaphragmatic injuries can be
missed and not repaired. Alternatively, inadequately
drained subphrenic bilious collections can cause an
00(0)
The American Surgeon 88(6)
Figure 2. (A) Intraoperative photo. Bronchobiliary fistula
extending through the diaphragm into the right lower lobe of
the lung. Black star: lung; black arrow: suture in the diaphragm
from original repair. (B) Surgical specimen. En bloc right lower
lobe wedge resection, bronchobiliary fistula, and portion of
diaphragm.
inflammatory reaction with subsequent erosion through
the diaphragm and extension into the bronchial system.1
A direct connection between the biliary system (under
positive intra-abdominal pressure) and the bronchial sys-
tem (under negative intrathoracic pressure) can create a
trans-diaphragmatic pressure gradient. This pressure gra-
dient promotes the back flow of bile into the bronchial
tree, potentiating the bronchobiliary fistula.2,3
Biliptysis is pathognomonic of bronchobiliary fistulas;
however, it is present in fewer than 50% of patients.1,4 More
often, patients present with vague symptoms such as fever,
chronic cough, or abdominal pain.1-3 Making the diagnosis
of bronchobiliary fistula is often difficult, especially in an
unstable trauma patient with multisystem injuries.
Additionally, the time from the inciting trauma to presenta-
tion varies, ranging from 2 to 73 days.1 Chest X-ray will
often show a right pleural effusion, atelectasis, or an ele-
vated right hemi-diaphragm.1-3 ACT scan may reveal a
pleural effusion or subphrenic fluid collection, but can miss
the diagnosis of bronchobiliary fistula.1-4 If there is a high
index of suspicion, pleural fluid and sputum should be eval-
uated for elevated bilirubin levels.1-3 Case reports have
shown that hepatobiliary scintigraphy (HIDA) scan is a
sensitive, noninvasive technique to visualize bile flow into
the airway or pleural cavity, confirming the diagnosis of
thoracobiliary fistula.1-3 However, positive pressure ventila-
tion reverses the trans-diaphragmatic pressure gradient;
thus, a HIDA scan can miss the diagnosis in an intubated
patient.3 Debate continues regarding the superiority of mag-
netic resonance cholangiopancreatography (MRCP) versus
ERCP in the diagnosis of bronchobiliary fistula. Although
MRCP is a safe, highly accurate, noninvasive imaging
modality with fewer complications than ERCP, ERCP can
identify the precise location of the bronchobiliary fistula
and offers decompressive therapeutic intervention, such as
sphincterotomy or biliary stent placement.4 Bronchoscopy
may reveal mucosal inflammation within the bronchus, but
Coughlin et al
it rarely visualizes the fistula; thus, it is unnecessary for
diagnosis.
Multiple management strategies have been described
in the literature; however, the optimal course has not been
well defined. Traditionally, the treatment has been pre-
dominantly surgical, but with recent advances in endos-
copy, more conservative approaches have come into
practice. Biliary decompression with ERCP and stent
placement or sphincterotomy has demonstrated success-
ful resolution of bronchobiliary fistulas in cases where
they were the result of iatrogenic injury, amebic or pyo-
genic liver abscesses, and biliary obstruction from cho-
ledocolithiasis.4 Although conservative measures have
proven to be successful in these instances, ERCP alone is
often inadequate in the setting of traumatic bronchobili-
ary fistula, with a nonoperative failure rate of 38%.1
Trauma patients with a bronchobiliary fistula can
develop worsening respiratory status from chemical
pneumonitis or are hemodynamically unstable from head
injuries, sepsis, or multisystem injury, which preclude
surgical intervention.3 In this case, ERCP may be utilized
to divert biliary flow away from the bronchial system,
allowing stabilization of the patient’s respiratory status
until surgical intervention is safe and feasible.2-4 Once the
patient is stable, or if conservative measures fail, surgical
intervention should not be delayed. Depending on the
underlying etiology of the bronchobiliary fistula, a thora-
cotomy, laparotomy, or thoracoabdominal incision can be
utilized.1-3 During thoracotomy, decortication is per-
formed, the fistulous tract is entirely excised, and after
inspecting the lung parenchyma, a wedge resection, seg-
mentectomy, or lobectomy is performed only if deemed
necessary.1-3 In addition, thoracotomy offers easy access
to the diaphragmatic defect, which can be repaired pri-
marily with a mesh, a pericardial fat pad, or a vascular-
ized intercostal muscle pedicle, depending on the size of
the defect and the tissue quality.1-3 During laparotomy,
subphrenic or intrahepatic fluid collections are drained,
liver injuries are repaired to ensure no further bile leak-
age, and Roux-en-Y hepaticojejunostomy or choledocho-
duodenostomy may be performed in order to relieve
persistent biliary obstruction. In their review of traumatic
bronchobiliary fistulas, Rothberg et al reported a 97%
success rate with surgical intervention.1
In the case that we present, there was low clinical sus-
picion of bronchobiliary fistula until the patient devel-
oped biliptysis, nearly one month after the inciting
trauma. Conservative management with biliary stenting
temporarily relieved his symptoms; however, he eventu-
ally required definitive surgical intervention, which
proved to be successful. In planning our operative
approach, the multiple surgical specialty teams took into
consideration the location of the prior hepatic injury and
3
1345
the hostile nature of his abdomen after evisceration and
several prior abdominal operations. We ultimately chose
a thoracic approach. This would allow for thorough
inspection of the lung, complete resection of the broncho-
biliary fistula, adequate drainage of the subphrenic space,
secure closure of the diaphragm, as well as the ability to
elevate the latissimus flap through the same incision. By
entering the thoracic cavity through the 8th intercostal
space, we were able to easily access the diaphragm and
superior dome of the liver, which avoided a difficult and
morbid transabdominal entry. In addition, we were able
to perform all three aspects of the operation through one
thoracic incision, which further reduced his morbidity.
Due to extensive injury to multiple segments of the right
lobe of the liver, there was significant concern that the
right lobe may not be salvageable. However, because we
accurately identified the entire fistulous tract, limited our
hepatic resection to segment seven, and provided ade-
quate perihepatic drainage with multiple drains, the
patient’s right lobe was successfully salvaged.
In conclusion, bronchobiliary fistulas are rare compli-
cations of thoracoabdominal trauma, which are associ-
ated with high morbidity and mortality. A high index of
suspicion in the appropriate clinical setting is crucial. In
the instance of a traumatic bronchobiliary fistula, surgical
intervention should be first-line therapy in order achieve
rapid resolution and a successful outcome.
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.
Funding
The author(s) received no financial support for the research,
authorship, and/or publication of this article.
References
1. Rothberg ML, Klingman RR, Peetz D, Ferraris VA, Berry WR.
Traumatic thoracobiliary fistula. Ann Thorac Surg.
1994;57(2):472-475. doi:10.1016/0003-4975(94)91022-7
2. Prodromos P, Condilis N. Thoracobiliary fistula. a rare
complication of thoracoabdominal trauma. Ann Ital Chir.
2009;80(6):467-470.
3. Nigro JJ, Arroyo H, Theodorou D, Velmahos GC, Bremner RM.
Bullets and biliptysis. Ann Thorac Surg. 2002;73(5):1645-1647.
doi:10.1016/S0003-4975(01)03464-6
4. Singh B, Moodley J, Sheik-Gafoor MH, Dhooma N,
Reddi A. Conservative management of thoracobiliary
fistula. Ann Thorac Surg. 2002;73(4):1088-1091. doi:10.
1016/S0003-4975(02)03382-9