Anda di halaman 1dari 18

Unusual Complications of

Minh B. Luu, MD*, Daniel J. Deziel, MD

 Gallstones  Complications  Lost stones  Fistula  Obstruction

 Extrinsic compression of the bile duct from gallstone disease is associated with bilio-
biliary fistulization requiring biliary-enteric reconstruction.
 Biliary-enteric fistulas are associated with intestinal obstruction at various levels. The pri-
mary goal of therapy is relief of intestinal obstruction; definitive repair is performed for
selected patients.
 Hemobilia from gallstone-related pseudoaneurysms is preferentially controlled by selec-
tive arterial embolization.
 Rapidly increasing jaundice with relatively normal liver enzymes is a diagnostic hallmark of
 Acquired thoraco-biliary fistulas are primarily treated by percutaneous and endoscopic in-


Gallstones are a routine commodity for general surgeons. Symptomatic disease pre-
sentations in their common forms of biliary colic, acute cholecystitis, choledocholithia-
sis, and gallstone pancreatitis are well known. This does not imply that these daily
conditions are without difficult challenges, even for the most experienced biliary sur-
geons. However, when stones migrate to involve adjacent viscera or vascular struc-
tures, the clinical challenge is far less familiar. Biliary eruption into the gut, into the
chest, or into the pelvis is a confounding situation; and, in circumstances that violate
major blood vessels, a rapidly catastrophic one. Most surgeons will encounter few, if
any, of these baffling patients during the course of their career. Yet, such occurrences
have been recognized for centuries and will continue. This article is a simple primer to
aid in the recognition and management of some of these gallstone oddities.

Department of General Surgery, Rush University Medical Center, Rush Medical College, 1633
West Congress Parkway, Chicago, IL 60612, USA
* Corresponding author.
E-mail address:

Surg Clin N Am 94 (2014) 377394
0039-6109/14/$ see front matter 2014 Elsevier Inc. All rights reserved.
378 Luu & Deziel


Biliary fistulas are abnormal communications of the biliary system to an internal or

external location. These conditions share a common pathophysiology of biliary
obstruction, infection, necrosis, and fistulization. Internal fistulas can form to the
gastrointestinal (GI) tract, thoracic cavity, genitourinary (GU) structures, or the vascular
system. External fistulas are communications of the biliary system to the skin either
spontaneously or by way of operatively lost stones. Bowel obstruction can result on
passage of large stones through bilioenteric fistulas with enlodgement in the small in-
testine or colon (gallstone ileus) or the pyloric region (Bouveret syndrome). Fistulization
between the gallbladder and extrahepatic bile ducts can also occur (Mirizzi syn-
drome). Although rare, these conditions may cause significant morbidity and mortality
that warrant attention.

Internal Fistula
Bilio-biliary fistula
Mirizzi syndrome
Background Although Hans Kehr1 in 1905 first reported on patients with partial bile
duct obstruction due to an impacted stone in the gallbladder, it was Pablo Luis Mirizzis
article in 1948 entitled Sindrome del conducto hepatico2 that called attention to this
Pathophysiology The eponym Mirizzi syndrome (MS) is used to describe mechan-
ical obstruction of the common hepatic duct due to extrinsic compression (with or
without an associated fistula) from stones in the gallbladder or cystic duct. The actual
obstruction is usually from a large impacted stone in the gallbladder neck or infundib-
ulum adjacent to the extrahepatic ducts (Fig. 1). Sometimes the extrinsic obstruction
is caused by a small stone impacted in the cystic duct that has a long parallel course to
the common hepatic duct or even by a tensely distended gallbladder itself. In his
famous article, Mirizzi2 incorrectly postulated a functional spasm of the common he-
patic duct as the cause of the bile duct obstruction. MS occurs in approximately 0.2%
to 1.5% of patients with gallstones. Beltran3 theorized that the external compression
of the bile duct and the later development of cholecystobiliary and cholcystoenteric
fistulas are different stages of the same disease process.
Classification Classification schemes are founded on the presence or absence of
fistulous erosion between the gallbladder and common bile duct and the extent of

Fig. 1. An intraoperative picture of a large stone removed from the infundibulum of a pa-
tient with Mirizzi syndrome.
Unusual Complications of Gallstones 379

common duct destruction. MS was classified by Mc Sherry and colleagues4 in 1982

into 2 types and reclassified by Csendes and colleagues5 in 1989 into 4 types. A
more recent modification added a fifth type that includes the presence of a bilioenteric
fistula with or without gallstone ileus (Fig. 2).6
Clinical presentation Patients with MS present in the fifth to seventh decade and are
usually women. A long-standing history of gallstone disease is usually elicited and the
presenting symptoms are those of obstructive jaundice. Additionally, patients with MS
also can present in the setting of acute cholecystitis, cholangitis, or pancreatitis.
Diagnosis Preoperative diagnosis of MS can be difficult, but careful surgical plan-
ning and intraoperative recognition may avoid the high incidence of bile duct injuries.7
Ultrasound and abdominal computed tomography (CT) are nonspecific but may be
suggestive when there is dilation of the proximal common hepatic or intrahepatic
ducts only. The accuracy of endoscopic retrograde cholangiopancreatography
(ERCP) to diagnose MS ranges from 55% to 90%. Because of the difficulty of preop-
erative diagnosis, more than one-half of MS cases are not diagnosed until operation.8
Characteristic findings of MS at surgery are fibrosis and obliteration of the Calot trian-
gle due to impacted stones in the infundibulum or cystic duct. The gallbladder may be
contracted or dilated with a thickened wall. Intraoperative cholangiography and ultra-
sonography are useful tools to delineate the abnormal anatomy in the midst of fibrosis
and inflammation (Fig. 3).
Treatment The goals of treatment for patients with MS are to decompress the biliary
tree and prevent recurrence. Temporary stenting of the bile duct during ERCP has

Fig. 2. Mirizzi syndrome type I to V. (A) Type I is the extrinsic compression of the common
bile duct by an impacted gallstone. (B) Type II involves one-third the circumference of the
bile duct. (C) Type III involves two-thirds the circumference of the bile duct. (D) Type IV
involves the whole circumference of the bile duct. (E) Type V involves types I to IV
with the addition of a bilioenteric fistula. (From Beltran MA, Csendes A, Cruces KS. The rela-
tionship of Mirizzi syndrome and cholecystoenteric fistula: validation of a modified classifi-
cation. World J Surg 2008;32:223743; with permission.)
380 Luu & Deziel

Fig. 3. A percutaneous transhepatic cholangiography showing a smooth external compres-

sion of the common hepatic duct due to a large stone in a patient with Mirizzi syndrome.

been used to allow time for surgical planning and medical optimization. Surgical treat-
ment is required and the extent based on the type of MS. Type I can be managed with
cholecystectomy or subtotal cholecystectomy alone, whereas types II, III, and IV often
require biliary reconstruction. Suggested surgical treatment based on Beltrans clas-
sification of MS is outlined in Table 1.9 Although the laparoscopic management of MS
has been described,10 surgeons should be wary because of the high risk of bile duct
injury. When diagnosed preoperatively, serious consideration should be given to
transferring the patient to a facility capable of major biliary reconstruction. Because
gallbladder cancer can be found in up to 25% of MS cases, an intraoperative frozen
section of the gallbladder wall should be performed and capability for an appropriate
oncologic resection available.11

Biliary-enteric fistula
Spontaneous internal biliary fistula: Most internal biliary fistulas are due to chronic or
neglected gallstone disease. Cholecystoduodenal fistulas are the most frequent, fol-
lowed by cholecystocolonic fistulas. Less common fistula sites are cholecystogastric,
cholecystocholedochal, choledochoduodenal, cholecystojejunal, and hepaticobron-
chial fistulas.1216 Choledochoduodenal fistulas are most commonly due to dorsal
erosion of a duodenal ulcer. A systematic review by Petrowsky and Clavien17 summa-
rized the distribution of biliary-enteric fistulas shown in Table 2.
Cholecystoduodenal fistulas account for 75% to 80% of cholecystoenteric fistulas.
Most are clinically silent or have vague digestive symptoms. Passage of large stones
can result in gallstone ileus. Cholecystocolic fistulas are the second most common
type.18 Costi and colleagues19 reviewed literature between 1950 and 2006 and found
231 cases. Only 7.9% of these cases were diagnosed preoperatively. In contrast to the
Unusual Complications of Gallstones 381

Table 1
New classification and suggested surgical treatment of Mirizzi syndrome

Type of
Mirizzi Description Treatment
I External compression of Open cholecystectomy
the bile duct Open subtotal cholecystectomy
Laparoscopic cholecystectomy
Laparoscopic subtotal cholecystectomy
IIa Cholecystobiliary fistula Open cholecystectomy
<50% the diameter of Open subtotal cholecystectomy
the bile duct
IIb Cholecystobiliary fistula Open subtotal cholecystectomy
>50% the diameter of Biliary-enteric derivation:
the bile duct Side-to-side to the duodenum
En-Y-de-Roux to the jejunum
IIIa Cholecystobiliary fistula and Simple closure of the fistula and treatment of
cholecystoenteric fistula the gallbladder according to the presence of
without gallstone ileus Mirizzi I, IIa, or IIb
IIIb Cholecystobiliary fistula and Treatment of the gallstone ileus and deferred
cholecystoenteric fistula treatment of the gallbladder according to the
with gallstone ileus presence of Mirizzi I, IIa or IIb

From Beltran MA. Mirizzi syndrome: history, current knowledge and proposal of a simplified clas-
sification. World J Gastroenterol 2012;18(34):463950.

benign presentations of most cholecystoduodenal fistulas, patients with fistulas to the

colon often report a sudden change from mild biliary symptoms to acute attacks. The
influx of bacteria from the colon into the biliary system is thought to be the cause of
the change in clinical condition. During the acute episode, most patients will have fe-
ver, chills, and abdominal pain. Nausea, weight loss, and diarrhea may be present due
to bile acid loss. In some circumstances, choleretic diarrhea caused by the irritant ef-
fect of bile acids on the colonic mucosa may be the primary symptom and indication
for surgery. Surgical treatment consisting of cholecystectomy with resection of the fis-
tula tract should be performed. Laparoscopic management has been described but
not widely used.20

Gallstone ileus
Background Gallstone ileus is classically described as the impaction of gallstone(s)
in the distal ileum and accounts for 1% to 3% of all mechanical bowel obstruction.21
The anatomist Thomas Bartholin first described gallstone ileus in 1654.22 In a series of
131 cases, Courvoisier,23 in 1890, reported a surgical mortality rate of 44%. More
contemporary mortality rates of 15% to 18% are still considerable and reflect the
associated afflictions of many patients with this condition.17,24,25
Clinical presentation Most cholecystoenteric fistulas are clinically silent, but if larger
(2 cm) stones enter the intestine, a mechanical obstruction results. Half of the pa-
tients with gallstone ileus will have a history of prior biliary symptoms and 20% to
30% of the patients have associated acute cholecystitis. Approximately 85% of
affected people are women and the median age is 70 years. Obstructive symptoms
may be partial and intermittent as the gallstone progresses aborally. This has been
referred to as a tumbling obstruction and may be responsible for the diagnostic
delay for several days that is characteristic in these patients. The Rigler triad, consist-
ing of small bowel obstruction (SBO), pneumobilia, and an aberrant stone in the GI
Luu & Deziel
Table 2
Incidence and frequency of biliary-enteric fistulas

Biliary-enteric Fistulas (%)

Author,Ref. year Incidence (%) Total Cholecystoduodenal Cholecystocolic Cholecystogastric Choledochoduodenal Others
Glenn, 1957 0.9 40 30 (76) 3 (7.5) 1 (2.5) 2 (5) 4 (10)
Half, 1971 0.4 24 13 (54) 4 (17) 1 (4) 4 (17) 2 (8)
Safaie, 1973 92 70 (76) 10 (11) 5 (5) 4 (4) 3 (3)
Patrassi, 1975 1.6 58 32 (55) 7 (12) 2 (3) 3 (5) 14 (24)
Yamashita, 1997 1.7 33 6 (18) 3 (9) 20 (61) 4 (12)
Total 1.0 247 151 (61) 27 (11) 9 (4) 33 (13) 27 (11)

From Petrowsky H, Clavien P. Biliary fistula, gallstone ileus, and Mirizzis syndrome. In: Clavien P, Baillie J, editors. Diseases of the gallbladder and bile ducts: diag-
nosis and treatment. 2nd edition. Malden (MA): Blackwell Publishing; 2006. p. 23951.
Unusual Complications of Gallstones 383

tract, is present in only 20% to 35% of reported cases.2628 Radiographic evidence of

pneumobilia should be carefully looked for, as it is present (sometimes only in retro-
spect) in most patients with gallstone ileus (Fig. 4). Its presence should tip off the pre-
operative diagnosis particularly when a mature woman presents with SBO without a
history of prior abdominal surgery or biliary tract intervention. Colonic obstruction,
usually the sigmoid, is often associated with a colonic stricture. CT (Fig. 5) has a sensi-
tivity of 93% to diagnose gallstone ileus, but the diagnosis is often delayed 3 to 8 days
after the onset of symptoms.29
Treatment Following adequate fluid and electrolyte resuscitation, surgery is indi-
cated. Relief of intestinal obstruction is the primary objective. The stone can usually
be gently manipulated proximal to the site of obstruction and removed by enterotomy
(Fig. 6). Concomitant definitive cholecystectomy and closure of the biliary-enteric fis-
tula is controversial. In most instances, operative treatment has been limited to relief of
obstruction; however, the potential for recurrent symptoms should prompt consider-
ation of definitive resection and repair for fit patients with residual stones. Reisner and
Cohen24 reported a higher mortality rate in a 1 stage (16.9%) compared with entero-
lithotomy alone (11.7%). Follow-up of those who underwent enterolithotomy alone
showed a 5% to 9% recurrence rate but only 10% of those who recurred required
reoperation. Spontaneous closure of the fistula may explain the low need for operative
intervention but is open to question. Marko Doko and colleagues29 also compared the
2 surgical management pathways and found that although mortality rates were similar,
patients who underwent index cholecystectomy had a higher rate of complications.
They concluded that cholecystectomy in the setting of gallstone ileus should be
reserved only for those with acute or gangrenous cholecystitis and residual stones.
If a cholecystectomy is done, the common bile duct must be evaluated for an obstruc-
tion that may have contributed to fistula formation. The surgeon can anticipate that
cholecystectomy in the setting of gallstone ileus will always be a more difficult oper-
ation. Interval cholecystectomy and fistula repair is considered for patients with resid-
ual stones or symptoms after enterolithotomy alone. In reality, this is practiced
infrequently because of the general medical status of many of these patients.

Fig. 4. Plain abdominal film demonstrating central pneumobilia and dilated small intestine
in gallstone ileus.
384 Luu & Deziel

Fig. 5. Coronal CT of an obstructing gallstone in the small intestine.

Bouveret syndrome Bouveret syndrome is a variant of gallstone ileus that was

described by Leon Bouveret in 189630 in 2 patients with gastric outlet obstruction
due to gallstones.31 The mechanism of obstruction is similar to gallstone ileus except
the site of obstruction is the duodenal bulb, postbulbar duodenum, pylorus, or pre-
pyloric area. Typical symptoms include nausea/emesis (86%) and abdominal pain
(71%). Other nonspecific signs and symptoms include anorexia, weight loss, abdom-
inal distension, and hematemesis. Endoscopic examination will often confirm gastric
outlet or duodenal obstruction (Fig. 7). Visualization of an impacted stone, however,
occurs in only 69% of cases because some stones can become deeply embedded
within the mucosa. CT is often used to confirm endoscopic findings and delineate
the anatomy of the biliary-enteric fistula. Despite the low success rate, endoscopic
retrieval of the obstructing stone should be attempted to minimize morbidity in a
high-risk patient population. Advances in endoscopic technology and instrumentation
should improve the success rate.32 Laser lithotripsy, mechanical lithotripsy, or extrac-
orporal shock wave lithotripsy have been tried. Operation by laparoscopic or open li-
thotomy (Fig. 8) with or without cholecystectomy and fistula closure is usually

Fig. 6. Intraoperative picture of an enterolithotomy in a patient with gallstone ileus.

Unusual Complications of Gallstones 385

Fig. 7. An endoscopic view of a gallstone obstructing the gastric outlet in a patient with
Bouveret syndrome.

necessary. Roux-en-Y duodenojejunostomy has been described to manage large


Biliary-vascular fistula
Hemobilia Hemorrhage into the biliary system was recognized by Francis Glisson in
1654 as a potential consequence of blunt or penetrating injury to the liver.34 Antoine
Portal, Professor of Anatomy and Surgery at College Royale de France, is credited
with the first ante mortem diagnosis of the condition in 1777 while he was treating a
servant of the Marquise de Cambis who suffered from hepatic abscesses.35 In the
modern era, Swedish surgeon Philip Sandblom made comprehensive study of this
problem in his 1972 monograph.36 It was he who coined the term hemobilia to
denote bleeding into or through the biliary tract.
Hemobilia may originate from the liver, the gallbladder, the intrahepatic or extrahe-
patic bile ducts, or from the pancreas (hemosuccus pancreaticus). Bleeding may
range from occult to massive and has a tendency to occur periodically. Clinically sig-
nificant hemobilia is usually from an arterial source, although venous bleeding can be
significant in cases of portal hypertension. Trauma is the most frequent antecedent of
hemobilia; either accidental trauma or, more commonly, iatrogenic trauma from any of
a litany of transhepatic or endoscopic interventions, including cholangiography,

Fig. 8. Intraoperative picture of an obstructing gallstone at the pylorus in a patient with

Bouveret syndrome.
386 Luu & Deziel

biopsy, drainage, stent placement, balloon dilatation, and lithotripsy. Other causes
include hepatobiliary malignancy, inflammatory conditions, vascular disorders, and,
of course, gallstones.
The symptoms of hemobilia depend on the rate of bleeding and the presence of bile
duct obstruction from clots. Flowing bile has natural fibrinolytic activity that dissolves
clots unless the bleeding is too rapid. Conversely, minor hemobilia may persist with
fibrinolysis and remain asymptomatic except for chronic anemia. The classic clinical
triad of biliary tract hemorrhage was highlighted by Quincke37 in 1871: gastrointestinal
hemorrhage, biliary colic, and jaundice. Gastrointestinal hemorrhage is most
commonly manifested as melena (90%), sometimes as hemetemesis (60%), with
biliary colic present in 70% of patients and jaundice in 60% of patients with clinically
apparent symptoms.36 Rarely, clot obstruction has caused acute cholecystitis or
pancreatitis.3844 There is a tendency to overlook the biliary tract as a source of signif-
icant gastrointestinal hemorrhage. Indeed, Professor Sandblom lamented that Blind
resection of a healthy stomach has been performed many times because of misinter-
preted hemorrhage into the biliary tract.36
Hemobilia from gallstones is usually microscopic and not uncommon. Fecal occult
blood has been demonstrated in one-quarter of patients with cholelithiasis.45 The
mechanism is direct trauma to the biliary mucosa from the stones. Gallstone disease
accounts for up to 10% of cases of major hemobilia.36,46 This occurs when gallstones
erode through the gallbladder wall or bile duct into an adjacent portion of the gut with
fistula formation and/or into an adjacent blood vessel with creation of a pseudoaneur-
ysm. Rupture of a pseudoaneurysm results in massive hemobilia, gastrointestinal
bleeding, and occasionally intraperitoneal bleeding as well.47 Not surprisingly, this
complication has frequently proven fatal.
Spontaneous gallstone erosion can involve various arteries, including the cystic ar-
tery, hepatic artery, replaced right hepatic artery (originating from the superior mesen-
teric artery), gastroduodenal artery, or even the aorta.36,4753 Pseudoaneurysms
resulting from operative trauma may cause life-threatening hemobilia as a late or early
complication following either open or laparoscopic cholecystectomy.5463 Post chole-
cystectomy pseudoaneurysms have been associated with bile leaks and formation of
late bile duct strictures.64 Three gallstones were discovered in the portal vein of Igna-
tius of Loyola at the time of his autopsy.65 Gallstones have been found in portal venous
collaterals along the common bile duct in a patient with portal vein thrombosis.36
When hemobilia is suspected, upper gastrointestinal endoscopy is the initial exam-
ination to exclude other sources and to directly visualize blood issuing from the
ampulla of Vater. Imaging by transabdominal ultrasonography, CT, magnetic reso-
nance imaging, or direct cholangiography via various routes can demonstrate blood
in the gallbladder or bile ducts and identify vascular anomalies. Selective hepatic arte-
rial angiography is the most specific method for identifying the source of an active
bleed and affords the opportunity for therapeutic embolization.
Treatment of gallstone-related hemobilia is by cholecystectomy and removal of bile
duct stones. When major arterial hemorrhage is identified, selective arterial emboliza-
tion is the mainstay of therapy today. Arterial injury discovered intraoperatively is
managed by vessel ligation or aneurysm repair. Bile duct obstruction caused by
hemobilia is successfully treated with endoscopic biliary drainage in most cases.66

Bilhemia Bilhemia is direct flow of bile into the blood through a biliary-vascular fistula.
This occurs when there is an abnormal communication between the higher-pressure,
typically obstructed, biliary system and the lower-pressure portal vein, hepatic veins,
or inferior vena cava. Clinically significant bilhemia is rare but has often resulted in fatal
Unusual Complications of Gallstones 387

pulmonary embolization of bile or sepsis.67 The cause is most often blunt trauma with
central liver rupture or iatrogenic penetrating injury associated with percutaneous
transhepatic interventions, such as biopsy, drainage, and placement of biliary stents
or transjugular intrahepatic porto-systemic shunts.6870 In exceptionally rare in-
stances, bilhemia may be caused by venous erosion of gallstones.36,65
The cardinal diagnostic feature of bilhemia is rapidly increasing jaundice and pro-
nounced direct hyperbilirubinemia without a substantial elevation of liver enzymes.68
Cholangiography by endoscopic retrograde or transhepatic approaches is the best
method for imaging the fistula. Unlike hemobilia, arteriography is not diagnostic. Hep-
atobiliary scintigraphy may also indicate the diagnosis.71
Treatment of bilhemia is founded on relief of bile duct obstruction by endoscopic,
transhepatic, or surgical means, which may result in spontaneous closure of the fistula.
Interruption of the fistula by placement of covered stents or by balloon tamponade or
angiographic coil occlusion has been successful.72,73 Although not applicable to gall-
stone bilhemia, operative solutions have included liver resection, liver transplantation,
and external drainage of the bile duct or disrupted liver.68

Biliary-thoracic fistula
Biliary-thoracic fistulas are abnormal communications between the biliary tract and
the thoracic cavity. The end point within the thoracic cavity can be either the pleural
space or the bronchial tree. The causes of biliary-thoracic fistulas are many and can
be categorized into thoracoabdominal trauma, parasitic liver disease, suppurative
biliary tract obstruction, or iatrogenic due to external drainage procedures. In Western
countries, gallbladder perforation, leading to subhepatic abscesses or postoperative
bile duct stenosis, is the primary cause of biliary-thoracic fistulas, whereas in devel-
oping countries, liver abscesses due to echinococcal or amebic infections are more
common.17 Patients often present with right upper quadrant and pleuritic pain, but
sometimes only vague symptoms, such as shortness of breath, are present.74 In pa-
tients with bronchobiliary fistulas, bitter taste and productive yellow sputum may be
present, suggesting bile pigments in sputum, known as biliptysis.75 Signs of systemic
infection, such as fever, chills, or leukocytosis, may be present in only half of the pa-
tients. Imaging will often show a right pleural effusion.76 Percutaneous and endo-
scopic interventions should first be attempted to treat acquired fistulas and surgery
should be reserved as second-line treatment.77 Singh and colleagues78 suggested a
treatment algorithm for thoracobiliary fistulas, as shown in Fig. 9.

Biliary-genitourinary fistula
Fistulous connections between the biliary tract and GU organs may occur spontane-
ously or consequent to accidental or operative trauma but are extremely unusual.
Connections have been described between the gallbladder or common bile duct
and the right renal pelvis, the urinary bladder, and the embryonic remnant ura-
chus.7982 Rabinowitz and colleagues82 reported on an elderly man with multiple ep-
isodes of urinary tract infections who was found to have gallstones in his urinary
bladder. Retrograde injection of a sinus at the dome of the bladder confirmed a
connection between the urachal remnant and the gallbladder. Definitive repair
involved resection of the gallbladder, bladder dome, and the urachal remnant. Fistu-
lization between the common bile duct and a neobladder ileal conduit has been suc-
cessfully managed endoscopically.83 Gallstone migration into GU structures has been
observed as a complication of gallstones lost at the time of cholecystectomy.8486
Symptoms include recurrent urinary tract infections and passage of gallstones
through the urethra. Gallstones in the urinary bladder may be amenable to cystoscopic
388 Luu & Deziel

Fig. 9. Suggested approach to the management of thoracobiliary fistulas. (From Singh B,

Moodley J, Sheik-Gafoor MH, et al. Conservative management of thoracobiliary fistula.
Ann Thorac Surg 2002;73(4):108891.)

extraction with spontaneous closure of the fistula tract. If the stones are multiple or
embedded in the bladder wall, operative removal may be necessary.
Although not specifically due to gallstone disease, a host of other rare urinary fis-
tulas have been described involving upper abdominal organs, including nephrohepatic
fistulas,87 pyelo-duodenal fistulas from pyonephrosis,88,89 and unusual fistulas from
migration of biliary stents.90 Posttraumatic nephro-bilary fistula has been treated by
percutaneous embolization.91
Spontaneous cholecystocutaneous fistula was once a common complication of
gallstone disease but now is a rare condition because of early diagnosis and treat-
ment. A total of 226 cases have been reported since the early twentieth century and
fewer than 25 cases have been reported in the past 50 years.92,93 Most patients are
women older than 50 years and most fistulas drain externally to the right upper quad-
rant. Other locations, such as chest wall, umbilicus, right groin, or gluteus, have been


Spillage of bile and gallstones during a laparoscopic cholecystectomy is not infre-

quent. The presence of cholecystitis, patient age, pigmented stones, number of
stones more than 15, and resident participation are risk factors for stone spillage.9698
Control of bile and stone spillage is not a major issue in open cholecystectomy, but
can be challenging laparoscopically. It is estimated that lost stones occur in 2% of
laparoscopic cholecystectomy cases with 8.5% of those cases resulting in subse-
quent complications.99 Although most lost stones are silent, they can cause wound
infection, abscess, or cutaneous sinus formation. Fistulization of lost stones can occur
internally to the GI tract or thoracic cavity or externally to the skin. Stones lost in the
Morrison pouch tend to migrate posteriorly and those lost in the gastrocolic omentum
may result in painful fibrotic masses. Fig. 10 is a list of complications reportedly due to
lost gallstones.99
Unusual Complications of Gallstones 389

Fig. 10. Diagram of possible complications of lost gallstones. (From Zehetner J, Shamiyeh A,
Wayand W. Lost gallstones in laparoscopic cholecystectomy: all possible complications. Am J
Surg 2007;193(1):738.)

Suggestions to minimize the spillage of stones and the resulting complications are
early controlled decompression of dilated gallbladders, early recognition and control
of the operation by the attending physician during a difficult case or with an inexperi-
enced trainee, and meticulous survey of the abdomen for spilled stones.
390 Luu & Deziel

The treatment of lost gallstone complications usually involves drainage of infection

and stone removal. Spontaneous closure of the fistula tract can be expected after
removal of gallstones.


The management of gallstone disease is commonplace for general surgeons. Yet, rar-
ities exist and, although not unique, their appearances may be seldom and so sepa-
rated in time as to elude recognition. The wary clinician who is cognizant of
anomalous biliary passages and stone migrations will be better prepared to handle
these unusual conditions that are benign, but potentially fatal and properly curable.


1. Kehr H. Die in neiner klinik geubte technik de gallenstein operationen, mit

einen hinweis auf die indikationen und die dauerersolge. Munchen (Germany):
JF Lehman; 1905.
2. Mirizzi P. Sindrome del conducto hepatico. J Int Chir 1948;8:7317.
3. Beltran MA. Mirizzi syndrome and gallstone ileus: an unusual presentation of
gallstone disease. J Gastrointest Surg 2005;9:6869.
4. Mc Sherry C, Ferstenberg H, Virhup M. The Mirizzi syndrome: suggested clas-
sification and surgical therapy. Surg Gastroenterol 1982;1:21925.
5. Csendes A, Diaz JC, Burdiles P, et al. Mirizzi syndrome and cholecystobiliary
fistula: a unifying classification. Br J Surg 1989;76:113943.
6. Beltran MA, Csendes A, Cruces KS. The relationship of Mirizzi syndrome and
cholecystoenteric fistula: validation of a modified classification. World J Surg
7. Lai EC, Lau WY. Mirizzi syndrome: history, present and future development. ANZ
J Surg 2006;76:2517.
8. Safioleas M, Stamatakos M, Safioleas P, et al. Mirizzi syndrome: an unexpected
problem of cholelithiasis: our experience with 27 cases. Int Semin Surg Oncol
9. Beltran MA. Mirizzi syndrome: history, current knowledge and proposal of a
simplified classification. World J Gastroenterol 2012;18:463950.
10. Yeh C, Jan Y, Chen M. Laparoscopic treatment for Mirizzi syndrome. Surg En-
dosc 2003;17:15738.
11. Redaelli CA, Buchler MW, Schilling MK, et al. High coincidence of Mirizzi syn-
drome and gallbladder carcinoma. Surgery 1997;121:5863.
12. Judd ES, Burden VG. Internal biliary fistula. Ann Surg 1925;81:30512.
13. Borman C, Rigler LG. Spontaneous internal biliary fistula and gallstone obstruc-
tion, with particular reference to roentgenologic diagnosis. Surgery 1937;1:
14. Puestow CB. Spontaneous internal biliary fistulae. Ann Surg 1942;115:104354.
15. Levowitz BS. Spontaneous internal biliary fistulas. Ann Surg 1961;154:24151.
16. Piedad OH, Wells PB. Spontaneous internal biliary fistula, obstructive and non-
obstructive types. Ann Surg 1971;175:7580.
17. Petrowsky H, Clavien P. Biliary fistula, gallstone ileus, and Mirizzis syndrome. In:
Clavien PA, Baillie J, editors. Diseases of the gallbladder and bile ducts: diagnosis
and treatment. 2nd edition. Malden (MA): Blackwell Publishing; 2008. p. 23951.
18. Antonacci N, Taffurelli G, Casadei R, et al. Asymptomatic cholecystocolonic fis-
tula: a diagnostic and therapeutic dilemma. Case Rep Surg 2013;2013:13.
Unusual Complications of Gallstones 391

19. Costi R, Randone B, Violi V, et al. Cholecystocolonic fistula: facts and myths. A
review of the 231 published cases. J Hepatobiliary Pancreat Surg 2009;16:818.
20. Gentileschi P, Forlini A, Rossi P, et al. Laparoscopic approach to cholecystocolic
fistula: report of a case. J Laparoendosc Surg 1995;5:4137.
21. Cooperman AM, Dickson ER, ReMine WH. Changing concepts in the surgical
treatment of gallstone ileus: a review of 15 cases with emphasis on diagnosis
and treatment. Ann Surg 1968;167:37783.
22. Deckoff SL. Gallstone ileus: a report of 12 cases. Ann Surg 1955;142:5265.
23. Courvoisier L. The pathology and surgery of the biliary tract. Leipzig (Germany):
FCW Vogel; 1890. p. 58.
24. Reisner RM, Cohen JR. Gallstone ileus: a review of 1001 reported cases. Am
Surg 1994;60:4416.
25. Moss JF, Bloom AD, Mesleh GF, et al. Gallstone ileus. Am Surg 1987;53:4248.
26. Balthazar EJ, Schechter LS. Air in gallbladder: a frequent finding in gallstone
ileus. AJR Am J Roentgenol 1978;131:21922.
27. Ripolles T, Miguel-Dasit A, Errando J, et al. Gallstone ileus: increased diagnostic
sensitivity by combining plain film and ultrasound. Abdom Imaging 2001;26:
28. Yu C, Lin C, Shyu R, et al. Value of CT in the diagnosis and management of gall-
stone ileus. World J Gastroenterol 2005;11:21427.
29. Marko Doko M, Zovak M, Kopljar M, et al. Comparison of surgical treatments of
gallstone ileus: preliminary report. World J Surg 2003;27:4004.
30. Bouveret L. Stenose du pylore adherent a la vesicule. Rev Med 1986;16:116.
31. Cappell MS, Davis M. Characterization of Bouverets syndrome: a comprehen-
sive review of 128 cases. Am J Gastroenterol 2006;101:213946.
32. Zhao J, Barrera E, Salabat M, et al. Endoscopic treatment for Bouveret syn-
drome. Surg Endosc 2013;27(2):655.
33. Erlandson MD, Kim AW, Richter HM III, et al. Roux-en-Y duodenojejunostomy in
the treatment of Bouveret syndrome. South Med J 2009;102:9635.
34. Glisson F. Anatomia hepatis. Amsterdam: J.J. Waesberge & E Weyerstraten; 1654.
35. Portal A. Observations sur la nature et le traitement des maladies du foie.
Charleston (SC): Caille et Ravier; 1813.
36. Sandblom P. Hemobilia (biliary tract hemorrhage). Springfield (MA): Charles C
Thomas; 1972.
37. Quincke H. Ein fall von aneurysma der leberarterie. Berl Klin Wochenschr 1871;
38. Counihan TC, Islam S, Swanson RS. Acute cholecystitis resulting from hemobilia
after tru-cut biopsy: a case report and brief review of the literature. Am Surg
39. Lee SL, Caruso DM. Acute cholecystitis secondary to hemobilia. J Laparoendosc
Adv Surg Tech A 1999;9:3479.
40. Parekh J, Corvera CU. Hemorrhagic cholecystitis. Arch Surg 2010;145:2024.
41. Van Os EC, Petersen BT. Pancreatitis secondary to percutaneous liver biopsy-
associated hemobilia. Am J Gastroenterol 1996;91:57780.
42. Wolf DS, Wasan SM, Merhav H, et al. Hemobilia in a patient with protein S defi-
ciency after laparoscopic cholecystectomy that caused acute pancreatitis: suc-
cessful endoscopic management. Gastrointest Endosc 2005;62:1636.
43. Kim JD, Lee KM, Chung WC, et al. Acute pancreatitis and cholangitis caused by
hemobilia from biliary papillomatosis. Gastrointest Endosc 2007;65:17780.
44. de Ribot X, Casellas F, Papo M, et al. Hemobilia causing acute pancreatitis after
percutaneous liver biopsy. J Clin Gastroenterol 1995;21:1712.
392 Luu & Deziel

45. Gad P. Okkult blodning ved galdesten. Nord Med 1962;68:1069.

46. Blumgart LH. Hemobilia and bilhemia. In: Surgery of the liver, biliary tract and
pancreas. 4th edition. Philadelphia: Saunders Elsevier; 2007. p. 106784.
47. Martinis A, Kalaitzis I, Basioukas P, et al. Vascular complications of large gall-
stones: proposal of a first classification. Hellenic J Surg 2012;84:3189.
48. England RE, Marsh PJ, Ashleigh R, et al. Case report: pseudoaneurysm of the
cystic artery: a rare cause of haemobilia. Clin Radiol 1998;53:725.
49. Bibyan M, Khandelwal RG, Reddy PK, et al. Gallstone causing pseudoaneur-
ysm of accessory right hepatic artery. Indian J Gastroenterol 2012;31:2134.
50. Broadbent NR, Taylor DE. Gallstone erosion of the aorta. ANZ J Surg 1975;45:
51. Siddiqui NA, Chawla T, Nadeem M. Cystic artery pseudoaneurysm secondary to
acute cholecystitis as cause of haemobilia. BMJ Case Rep 2011;2011. pii:
52. Wang S, Wang X. Hemobilia secondary to acute gangrenous cholecystitis. Am
Surg 2012;78:14951.
53. Priya H, Anshul G, Alok T, et al. Emergency cholecystectomy and hepatic arte-
rial repair in a patient presenting with haemobilia and massive gastrointestinal
haemorrhage due to a spontaneous cystic artery gallbladder fistula
masquerading as a pseudoaneurysm. BMC Gastroenterol 2013;13:43.
54. Larmi TK. Hemobilia associated with cholecystitis, postcholecystectomy condi-
tions and trauma: review of 12 cases. Ann Surg 1966;163:37381.
55. Zilberstein B, Cecconello I, Ramos AC, et al. Hemobilia as a complication of
laparoscopic cholecystectomy. Surg Laparosc Endosc 1994;4:3013.
56. Stewart BT, Abraham RJ, Thomson KR, et al. Post-cholecystectomy haemo-
bilia: enjoying a renaissance in the laparoscopic era? Aust N Z J Surg
57. Saldinger PF, Wang JY, Boyd C, et al. Cystic artery stump pseudoaneurysm
following laproscopic cholecystectomy. Surgery 2002;131:5856.
58. Dogru O, Cetinkaya Z, Bulbuller N, et al. Hemobilia: a rare complication of lapa-
roscopic cholecystectomy. Surg Endosc 2003;17:14956.
59. Stewart L, Robinson TN, Lee CM, et al. Right hepatic artery injury associated
with laparoscopic bile duct injury: incidence, mechanism, and consequences.
J Gastrointest Surg 2004;8:52330.
60. Srinivasaiah N, Bhojak M, Jackson R, et al. Vascular emergencies in cholelithi-
asis and cholecystectomy: our experience with two cases and literature review.
Hepatobiliary Pancreat Dis Int 2008;7:21720.
61. Yao CA, Arnell TD. Hepatic artery pseudoaneurysm following laparoscopic cho-
lecystectomy. Am J Surg 2010;199:101.
62. Hadj AK, Goodwin M, Schwalb H, et al. Pseudoaneurysm of the hepatic artery.
J Gastrointest Surg 2011;15:1899901.
63. Petrou A, Brenna N, Soonawalla Z, et al. Hemobilia due to cystic artery stump
pseudoaneurysm following laparoscopic cholecystectomy: case presentation
and literature review. Int Surg 2012;97:1404.
64. Madanur MA, Battula N, Sethi H, et al. Pseudoaneurysm following laparoscopic
cholecystectomy. Hepatobiliary Pancreat Dis Int 2007;6:2948.
65. Ullman WH. Autopsy of Ignatius of Loyola. Monogr Soc Res Child Dev 1963;35:
66. Kim KH, Kim TN. Etiology, clinical features, and endoscopic management of he-
mobilia: a retrospective analysis of 37 cases. Korean J Gastroenterol 2012;59:
Unusual Complications of Gallstones 393

67. Sandblom P, Jakobsson B, Lindgren H, et al. Fatal bilhemia. Surgery 2000;127:

68. Glaser K, Wetscher G, Pointner R, et al. Traumatic bilhemia. Surgery 1994;116:
69. Brown CY, Walsh GC. Fatal bile embolism following liver biopsy. Ann Intern Med
70. Mallery S, Freeman ML, Peine CJ, et al. Biliary-shunt fistula following transjugular
intrahepatic portosystemic shunt placement. Gastroenterology 1996;111:13537.
71. Francois D, Walrand S, Van Nieuwenhuyse JP, et al. Hepatobiliary scintigraphy
in a patient with bilhemia. Eur J Nucl Med 1994;21:10203.
72. Struyven J, Cremer M, Pirson P, et al. Posttraumatic bilhemia: diagnosis and
catheter therapy. AJR Am J Roentgenol 1982;138:7467.
73. Rankin RN, Vellet DA. Portobiliary fistula: occurrence and treatment. Can Assoc
Radiol J 1991;42:559.
74. Cunningham L, Grobman M, Paz H, et al. Cholecystopleural fistula with choleli-
thiasis presenting as a right pleural effusion. Chest 1990;97:7512.
75. Gugenheim J, Ciardullo M, Traynor O, et al. Bronchobiliary fistulas in adults. Ann
Surg 1988;207:904.
76. Delco F, Domenighetti G, Kauzlaric D, et al. Spontaneous biliothorax (thoraco-
bilia) following cholecystopleural fistula presenting as an acute respiratory insuf-
ficiency: successful removal of gallstones from the pleural space. Chest 1994;
77. Poullis M, Poullis A. Biliptysis caused by a bronchobiliary fistula. J Thorac Car-
diovasc Surg 1999;118:9712.
78. Singh B, Moodley J, Sheik-Gafoor MH, et al. Conservative management of thor-
acobiliary fistula. Ann Thorac Surg 2002;73:108891.
79. Op den Orth JO. A radiologically demonstrated fistula between the common bile
duct and the right renal pelvis. Radiol Clin Biol 1969;38:4025.
80. Holst S, Peterson NE, McCroskey BL. Pyelo-choledochal fistula accompanying
operative-cholangiography. J Urol 1988;139:8234.
81. Shah A, Salamut W, Rull G, et al. Spontaneous pelvi-cholecystic fistula. BJU Int
82. Rabinowitz CB, Song JH, Movson JS, et al. Cholecysto-urachal fistula. Abdom
Imaging 2007;32:10810.
83. Narayan S, Roslyn J, Raz S, et al. Successful endoscopic treatment of a fistula
between the common bile duct and neobladder. Gastrointest Endosc 1993;39:
84. Castro MG, Alves AS, Oliveira CA, et al. Elimination of biliary stones through the
urinary tract: a complication of laparoscopic cholecystectomy. Rev Hosp Clin
Fac Med Sao Paulo 1999;54:20912.
85. Lutken W, Berggren P, Maltbak J. Passing of gallstones via the urethra: a compli-
cation of laparoscopic cholecystectomy. Surg Laparosc Endosc 1997;7:4957.
86. Famulari C, Pirrone G, Macri A, et al. The vesical granuloma: rare and late
complication of laparoscopic cholecystectomy. Surg Laparosc Endosc Percu-
tan Tech 2001;11:36871.
87. Abraham JI, Martin JL, Smith EH. Nephrohepatic fistula. N Y State J Med 1971;
88. Boardman KP. Duodenal and common bile duct obstruction with a pyelo-
duodenal fistula caused by renal calculous pyonephrosis. Br J Urol 1976;48:6.
89. Desmond JM, Evans SE, Couch A, et al. Pyeloduodenal fistulae: a report of two
cases and review of literature. Clin Radiol 1989;40:26770.
394 Luu & Deziel

90. Basile A, Macri A, Lamberto S, et al. Duodenoscrotal fistula secondary to retro-

peritoneal migration of an endoscopically placed plastic biliary stent. Gastroint-
est Endosc 2003;57:1368.
91. Ryu R, Novak Z, Coldwell D. Percutaneous embolization of a posttraumatic
nephrobiliary fistula. J Trauma 1998;44:38991.
92. Yuceyar S, Erturk S, Karabicak I, et al. Spontaneous cholecystocutaneous fistula
presenting with an abscess containing multiple gallstones: a case report. Mt Si-
nai J Med 2005;72:4024.
93. Sayed L, Sangal S, Finch G. Spontaneous cholecystocutaneous fistula: a rare
presentation of gallstones. J Surg Case Rep 2010;2010:5.
94. Malik AH, Nadeem M, Ockrim J. Complete laparoscopic management of chol-
ecystocutaneous fistula. Ulster Med J 2007;76:1667.
95. Nicholson T, Born MW, Garber E. Spontaneous cholecystocutaneous fistula pre-
senting in the gluteal region. J Clin Gastroenterol 1999;28:2767.
96. Woodfield J, Rodgers M, Windsor J. Peritoneal gallstones following laparo-
scopic cholecystectomy. Surg Endosc 2004;18:12007.
97. Barrat C, Champault A, Matthyssens L, et al. Iatrogenic perforation of the gall-
bladder during laparoscopic cholecystectomy does not influence the prognosis:
prospective study. Ann Chir 2004;129:259.
98. Brockmann J, Kocher T, Senninger N, et al. Complications due to gallstones lost
during laparoscopic cholecystectomy. Surg Endosc 2002;16:122632.
99. Zehetner J, Shamiyeh A, Wayand W. Lost gallstones in laparoscopic cholecys-
tectomy: all possible complications. Am J Surg 2007;193:738.