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220 J. Robert O’Neill and R.W. Parks
cholangitis and biliary obstruction can lead to cirrhosis and portal hypertension, and
patients should be thoroughly assessed for these complications prior to operative
intervention [13, 15, 16].
Up to 75% of patients with type I cysts have ductal stones, and the prevalence is
even higher in patients with type IVa cysts [17]. It is important to establish the
extent of intrahepatic disease and the presence of abscesses, stones, or strictures
pre-operatively, as these pathologies may not be adequately treated by hepaticojejunostomy and may result in recurrent stone formation and sepsis [17].
Patients with choledochal cysts have up to a 100-fold higher risk of developing
cholangiocarcinoma than the general population [18]. The incidence of malignancy
increases with age, with the majority of cancers presenting by the fourth decade of
life. The overall incidence of malignancy is 11%, [2, 19, 20] and those patients with
type I or type IVa cysts appear to be at highest risk [21, 22].
The risk of cholangiocarcinoma remains following resection of extrahepatic
cystic disease, with a 10% reported cumulative incidence in one Japanese series up
to 25 years post-excision, although the risk appears lower in Western populations
[18, 23]. Patients at higher risk include those with an APBJ or those with incomplete cyst excision, such as patients with type IVa cysts and residual intrahepatic
disease [22].
A key principle in the treatment of choledochal cysts is complete excision of the
extrahepatic disease, as patients who have undergone a cyst drainage procedure—
such as transduodenal sphinctero plasty, choledochoduodenostomy, or choledochojejunostomy—have a higher risk of cholangiocarcinoma than those patients who
have undergone cyst excision [23, 24].
Interestingly, drainage procedures in patients without choledochal cysts are also
associated with an increased risk of cholangiocarcinoma, and this may reflect the
carcinogenic effect of chronic enteric reflux on the biliary epithelium [25].
Alternative Approaches and Controversies
• Patients with Type IVa choledochal cysts have both intra and extrahepatic
disease. One approach is to only resect the extrahepatic disease. An
alternative is to resect both the intrahepatic and extrahepatic components,
with a greater risk of post-operative morbidity but potentially a reduced
risk of future intrahepatic complications.
• Our approach is to undertake excision of the extrahaptic disease only,
reserving synchronous resection for selected patients with predominantly
unilobar disease and evidence of intrahepatic strictures or multiple intrahepatic stones at high risk of recurrent biliary sepsis.
16 Surgical Resection of a Type IVa Choledochal Cyst 221
Operative Management
Our patient underwent laparotomy with intraoperative ultrasonography, confirming
dilated intrahepatic and extrahepatic ducts with no evidence of ductal stones, liver
abscess, or other parenchymal lesions. The extrahepatic biliary tree was excised
from the confluence of the hepatic ducts to the superior border of the pancreas with
an en-bloc cholecystectomy. On-table choledochoscopy was perfor med and confirmed no intrahepatic ductal stones or strictures (See Technical Elements box
below).
A Roux-en-Y hepaticojejunostomy was performed for reconstitution of biliary
drainage. This would be our favoured approach for both type I and type IVa
choledochal cysts, and is increasingly recognised as the established standard in the
literature [26]. Cyst excision and reconstruction should be performed in all cases,
including those patients who have previ ously undergo ne a primary cyst drainage
procedure, as post-operative complication rates are low and longer term outcomes
are superior [24]. If the gallbladder is present, a cholecystectomy should always be
performed as part of the procedure [27].
Our practice would be to perform the biliary-enteric anastomosis at the confluence of right and left hepatic ducts. Spatulation of the left hepatic duct allows a
wide anastomosis to be constructed in most patients, reducing the risk of a
post-operative anastomotic stricture and therefore reducing the need for revisional
surgery [28]. If the intrahepatic component of the choledochal cyst involves the
confluence of the hepatic ducts, we would still recommend excision of the extrahepatic component and anastomosis to an epithelial-lined portion of the intrahepatic
cyst. In patients with distal extrahepatic disease, complete excision of the extrahepatic ducts and anastomosis at the biliary confluence should still be performed.
The feasibility of laparoscopic resection and reconstruction for a Type IVa
choledochal cyst has been reported with outcomes from case series comparable to
those after open surgery [29]. However, previous recurrent attacks of cholangitis
and pancreatitis can make these cases particularly challenging, and this approac h
should only be attempted by surgeons with advanced laparoscopic skills and
experience in complex hepatobiliary surgery.
Partial hepatectomy to excise the intrahepatic cystic disease has been proposed
for patients with type IVa choledochal cysts [30–32]. Advocates of this approach
suggest there are fewer long-term complications with intrahepatic stone disease and
cholangitis, [2] however, the higher post-operative morbidity rates have been cited
as justification for a more conservative approach. The optimal approach remains an
area of controversy, with only case series to guide recommendations [33, 34].
Our approach would be to consi der synchronous hepatic resection and extrahepatic cyst excision if the intrahepatic disease is predominantly unilobar, with a
dominant intrahepatic ductal stricture or extensive intrahepatic stones, either of
which may predispose to cho lestasis and recurrent cholangitis. Hepatic resection
should be anatomical and undertaken as for other indications, preserving sufficient
222 J. Robert O’Neill and R.W. Parks
future liver remnant volume, adjusting for underlying liver disease if biliary cirrhosis is suspected.
Outcome
Our patient made an uncomplicated post-operative recovery with no septic complications, and was discharged on the fifth post-operative day with normal liver
function tests. She remains well, with normal liver function at last follow-up, 3
years after her procedure.
Post-operative cholan gitis has been reported after primary excision of the
extrahepatic portion of Type IVa cysts [2, 28]. The risk of post- operative complications, specifically cholang itis or anastomotic stricture, appears higher in patients
with previous surgery, especially cyst drainage procedures.
Due to the risk of post-operative stricture, stone formation, and development of
cholangiocarcinoma, it is recommended that patients should undergo routine
follow-up [ 33 , 35]. Cases of cholangiocarcinoma have been reported over 30 years
after the primary resection of a choledochal cyst, and therefore this monitoring
should continue lifelong [18, 36]. Patients with choledochal cysts are at risk of
developing malignancy throughout their biliary tree, and this risk, although lower,
persists post-operatively. Therefore, even those patients with type I cysts who have
undergone complete cyst excision should remain under surveillance [37].
It is unclear from published series what the optimal follow-up interval should be
and whether serial imaging is required to detect complications. It is also not clear if
follow-up improves outcome, as the median survival in a series of 32 patients who
developed cholangiocarcinoma during follow-up after cyst excision was just
15 months [18].
In the absence of evidence, our pragmatic approach is to follow patients who are
otherwise well by annual clinical review and routine blood tests. If patients become
symptomatic or liver function tests become abnormal, then an MRCP is undertaken
to identify evidence of intrahepatic stones, strictures, abscesses or pancreaticobiliary malignancy.
Technical Elements
• Pre-operative imaging is essential to plan the extent of resection and
identify aberrant anatomy.
• Dissection can be challenging, as most adult patients will have had previous episodes of pancreatitis, cholangitis and/or operative drainage
procedures.
• Intraoperative ultrasound should be used to con firm the distribution of
cystic disease and ductal stones, and can identify occult intrahepatic
masses or abscesses.
16 Surgical Resection of a Type IVa Choledochal Cyst 223
• During resection the hilar plate should be lowered to allow access to the
confluence of hepatic ducts. Our standard approach would be to only
excise the extrahepatic cyst, and this would generally denote the superior
limit of the resection.
• Intraoperative choledochoscopy should be performed to confirm there are
no residual intrahepatic stones or strictures.
• Reconstruction with a Roux-en-Y hepaticojejunostomy at the biliary
confluence should be performed to minimise the risk of post-operative
stricture. Our favoured method is to perform an end-to-side, single layer,
mucosa-to-mucosa, hepaticojejunostomy using interrupted 4/0 polydioxanone (PDS
®
) suture.
• Some surgeons advocate a hepaticoduodenostom y for reconstruction,
predominantly in the paediatric setting. This offers the advantage of
providing endoscopic access to the biliary tree.
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Bile Duct Injury at the Hepatic Confluence
Miguel-Angel Mercado, Mario Vilatoba
and Bruno A. Gonzalez-Nolasco
Clinical Case
A 32-year-old female with history of biliary colic underwent a cholecystectomy
with no incidents reported. During the third postoperative day, abdominal pain and
distention were noted. An abdominal ultrasound showed free abdominal fluid. An
exploratory laparoscopic procedure was performed, showing bile in the abdominal
cavity, which was drained accordingly, and several surgical drains were left in the
cavity.
An endoscopic retrograde cholangiopancreatography (ERCP) was performed,
which showed complete transection of the common hepatic duct. The right upper
abdominal drain showed continuous drainage between 500 and 750 ml a day.
The patient was referred to our center on the eighth postoperative week. Upon
physical examination, the patient was in good general condition , without signs of
peritoneal irritation or jaundice. The abdomen was flat with adequate peristalsis.
A drain on the right upper quadrant continued to actively drain bile. A magnetic
resonance cholangiopancreatography (MRCP) was performed (Fig. 17.1), showing
a biliary injury classified as Strasberg E-4; therefore, no attempts to place a percutaneous drainage were done, and the patient was scheduled for surgical repair.
Surgical treatment of bile duct injury is indicated when loss of duct continuity is
found and an endoscopic and/or radiological approach is ruled out [1]. Roux-en-Y
hepaticojejunostomy has been proven to be the best treatment option by several
groups [2–5]. A high-quality bilioenteric anastomosis, defined as a tension-free,
wide, with adequate suture material, done in healthy, non-scarred non-ischemic
17
M.-A. Mercado (&) M. Vilatoba B.A. Gonzalez-Nolasco
Department of General Surgery, National Institute of Medical Sciences and Nutrition
“Salvador Zubiran”, Vasco de Quiroga no. 15, Col. Seccion XVI, Tlalpan,
Mexico City 14080, Mexico
e-mail: mercadiazma@yahoo.com
© Springer International Publishing AG 2017
T.M. Pawlik et al. (eds.), Case-Based Lessons in the Management of Complex
Hepato-Pancreato-Biliary Surgery, DOI 10.1007/978-3-319-50868-9_17
227
228 M.-A. Mercado et al.
Fig. 17.1 MRI showing loss of confluence.
ducts that are anastomosed to a defunctionalized Roux-en-Y jejunal limb, offers the
best results [6]. There are several technical maneuvers that can be done in order to
reach this goal, including the anterior opening of the confluence and the left duct, as
well as partial removal of hepatic segments IV and V [7].
Our group has shown that an anastomosis performed in a patient with preserved
confluence offers the best results [8]. These results can also be optimized if the
patient has no stones or biliary sludge, which are usually developed as a result of
bacterial colonization.
Loss of confluence, classified as a Bismuth IV [9] or a Strasberg E-4, [3]isa
technical challenge for the surgeon. This is also, in our experience, the most
undesirable scenario for repair and long-term results are unpredictable. In some
cases, the anatomical variation of a low confluence results in a higher rate of bile
duct injury [10]. After section and ablation of the duct, two separated lumens can be
observed.
In other situations, ischemic damage due to thermal energy may be the cause of
the injury, secondary to the heuristic error in which the common bile duct is
mistaken with the cystic duct. Also, the presence of a biloma (infected or not), as
well as the anatomical deformity contribute to the ductal damage. The most common cause for biliary injury in our series is a technically deficient repair attempt,
usually performed by the primary-care surgeon.
17 Bile Duct Injury at the Hepatic Confluence 229
Intraoperative Technical Pearls
• Nearby organs should be methodically separated and, in patients who
have previously undergone biliodigestive derivation, it is particularly
important to free the small intestine in order to determine whether the
anastomosed loop is not obstructed or defunctionalized, in case there is an
enteral anastomotic variant (Omega loop with Braun anastomosis,
Nakayama Beta-anastomosis), or if there is an abnormal positioning of the
loop compromising its appropriate function.
• In our center’s experience, longitudinally sectioning the anterior aspect of
the duct (considering circulation is located on the lateral aspects) and
directing this section toward the left duct without moving its posterior
aspect makes creating the confluence a simpler task. Every section on the
anterior aspect measures approximately 2–3 mm, having thoroughly
verified and certified the direction of the ducts.
• In order to expose the hepatic hilum, the base of segment IV is removed
with a 3 3 3 cm wedge. The small parenchymal vessels bleeding are
controlled mainly through compression and, in some cases, using transfictive 5-0 sutures. This maneuver adequately exposes the left duct that
follows an extrahepatic trajectory, from the confluence to the round
ligament’s end.
The loss of confluence can be easily diagnosed nowadays with the aid of
MRCP. Endoscopic management can be a suitable therapeutic procedure by placing
a percutaneous biliary drain or stents in order to maintain the function of the ducts.
When endoscopic treatment is not an option, the surgical alternatives available for
complex biliary injuries are: Portoenterostomy, double barrell anastomosis, construction of a neoconfluence, partial hepatectomy, and liver transplant ation.
Portoenterostomy
This is the adult variant of the Kasai procedure [11]. It is the least desired option
hence it has presented a high failure rate in our center [12]. We suggest its usage
when very small, joined ducts are found and the construction of a high-quality
bilioenteric anastomosis (wide, tension free, with appropriate epithelization of
mucosae, done in healthy ducts using adequate sutur e material) is not feasible. In
some cases, it is possible to place percutaneous stents during the preoperative or
postoperative period in order to advance them to the intestinal lumen at the time of
portoenterostomy. Along with periodical changes of the percutaneous stents, this
option allows the patient to maintain an acceptable quality of life (without jaundice
and cholangitis, with the evident disadvantage of having an indwelling catheter for
a long period of time).
230 M.-A. Mercado et al.
When the stents are removed, failure of the patency of the ducts is almost
constant. In our hands, several of these cases are enrolled into the liver transplant
waiting list.
Double Barrell Anastomosis
This variant can be performed when the ducts are widely separated (more than
1 cm). The right duct anastomosis is technically demanding and it anticipates a high
chance of long-term dysfunction. Even after stenting, the final outcome after
removal is unpredictable.
The anastomosis to the left duct can usual ly be done with a moderate level of
difficulty by extending the incision to the anterior aspect of the duct. We have less
than 10 cases repaired with this surgical approach. Complications such as secondary biliary cirrhosis may arise (with or without cholangitis, which in some
conditions is severe). A couple of cases in our series have been treated by means of
unilobar portal vein embolization with the objective of inducing atrophy of the
affected liver lobe. Segmentary portal vein embolization does not offer good results.
Alternative Approaches and Controversies
• There are unfortunate isolated cases in which an appropriate anastomosis
is impossible to perform. The anastomosis of the jejunal opening to the
hepatic parenchyma and the need of stents in every duct included in the
anastomosis technically leads to a portoenterostomy, similar to the one
described by Kasai [13]. In our experience and that of others, these are
very infrequent cases that often require prolonged stenting, and may also
develop acute cholangitis episodes, and thus should be considered for
liver transplant.
• Endoscopic access is not a simple procedure in these types of patients
since it is challenging to insufflate the intestinal loop. If this is feasible,
there is also the problem of identifying the anastomosis that oftentimes is
punctiform and/or obstructed.
• There are numerous reports that have discussed the use of hepaticoduodenal derivation to successfully repair the biliary tract, such as Traverso’s
and Stewart–Way’s groups; [14] it has been recommended for injuries in
which the ductus choledochus is of acceptable length and there is no loss
of tissue. From our viewpoint, it has the disadvantage of exposing the
anastomosis to acidic content, and food and vegetable residues that hinder
the anastomosis’ function. Another disadvantage of this type of derivation
is that if early dehiscence develops, aside from a biliary fistula, a duodenal
fistula also appears with disastrous short- and long-term consequences.