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J Gastrointest Surg

DOI 10.1007/s11605-014-2530-4

ORIGINAL ARTICLE

Surgical and Endoscopic Management of Remnant Cystic


Duct Lithiasis After Cholecystectomy—a Case Series
Michael R. Phillips & Mark Joseph & Evan S. Dellon &
Ian Grimm & Timothy M. Farrell & Christopher C. Rupp

Received: 18 December 2013 / Accepted: 21 April 2014


# 2014 The Society for Surgery of the Alimentary Tract

Abstract
Introduction Postcholecystectomy syndrome (PCS) as a result of remnant cystic duct lithiasis (RCDL), or gallstones within the
cystic duct after cholecystectomy, can cause persistent or recurrent symptoms after cholecystectomy.
Study Design A retrospective descriptive analysis was performed for all patients with RDCL at a single institution between 2001
and 2012. Details of presentation, diagnosis, and surgical and endoscopic treatments, and outcomes were collected and analyzed.
Results Twelve patients with RCDL were identified. The interval between cholecystectomy to RCDL discovery was 34.2 months
(range 0.5–168 months). On a standard liver enzyme panel, 75 % of patients had derangements in ≥1 indices, with the most
common single laboratory test abnormality occurring in gamma-glutamyl transferase (GGT) (80 %). Eight operative reports
noted that the cystic duct was noticeably dilated at the time of cholecystectomy. Two patients developed a cystic duct leak
(Strasberg type A bile duct injury) postoperatively, which was managed nonoperatively. Six cases of RCDL required surgery, and
six were managed endoscopically.
Conclusion RCDL is a potential cause of postcholecystectomy syndrome, but the true incidence is unknown. Laboratory
analysis and imaging are helpful in establishing the diagnosis of RCDL. Endoscopic therapy has a role in the treatment of
RCDL, but surgical excision of the remnant cystic duct lithiasis may be required.

Keywords Remnant cystic duct lithiasis (RCDL) .


Cholecystectomy . Postcholecystectomy syndrome

Abbreviations Introduction
RCDL Remnant cystic duct lithiasis
GGT Gamma-glutamyl transferase Cholecystectomy is the most common abdominal operation
PCS Postcholecystectomy syndrome performed by general surgeons, with an estimated 750,000
AST Aspartate aminotransferase cases in the USA each year.1 Most cholecystectomies
ALT Alanine aminotransferase are performed due to the sequelae of cholelithiasis, with
the vast majority of patients experiencing complete
M. R. Phillips (*) : M. Joseph : T. M. Farrell
symptom relief after cholecystectomy. However, approx-
Department of Surgery, University of North Carolina at Chapel Hill, imately 5 % of patients continue to have symptoms
101 Manning Drive, Chapel Hill, NC 27514, USA after cholecystectomy and are sometimes designated as

e-mail: miphilli@unch.unc.edu suffering from postcholecystectomy syndrome (PCS).2 6
E. S. Dellon : I. Grimm
It has been suggested that the majority of patients who
Division of Gastroenterology and Hepatology, Department of develop PCS actually suffer from nonbiliary disorders, such
Medicine, University of North Carolina at Chapel Hill, 101 Manning as gastroesophageal reflux disease, peptic ulcer disease,
Drive, Chapel Hill, NC 27514, USA nonulcer (functional) dyspepsia, or chronic pancreatitis.5
However, a small percentage of patients truly have symptoms
C. C. Rupp
Department of Surgery, Prevea Health, 1715 Dousman Street, Green attributable to the extrahepatic biliary tree, from etiologies
Bay, WI 54303, USA such as biliary strictures, retained choledocholithiasis,
J Gastrointest Surg

sphincter of Oddi dysfunction, and remnant cystic duct lithi- Results


asis (RCDL).
The etiology of true RCDL is presumed to be from calculi A total of 12 patients (5 men, 7 women) with RCDL were
remaining from the initial cholecystectomy, rather than calculi identified. The mean age at presentation for RCDL was
developing de novo because of a subtotal cholecystectomy. 38.3 years (range 24–57 years), and average body mass index
Estimates suggest that at the time of cholecystectomy, nearly was 35.2 kg/m2. The mean interval from initial cholecystec-
15 % of patients have lithiasis present in the cystic duct, which tomy to discovery of RCDL was 34.2 months (range 0.5–168
has been shown to correlate preoperatively with choledocho- months). Final pathologic diagnoses at the initial cholecystec-
lithiasis, elevated liver enzymes, and pain in the months tomy included chronic cholecystitis (seven), acute cholecysti-
preceding cholecystectomy. Some series have reported that tis (four), and gangrenous cholecystitis (one). Only four of the
up to 30 % of patients suffering from PCS have RCDL as the patients had their initial surgery at our institution, and opera-
,
etiology of persistent symptoms.7 8 tive procedures performed included eight laparoscopic chole-
The true incidence of retained calculi in the cystic duct cystectomies (one with intraoperative cholangiogram) and
stump, however, has not been described. Most published two open cholecystectomies. The reasons for conversion to
series are small and aggregate heterogeneous disease process- an open procedure or the omission of intraoperative cholan-
es, including patients with remnant gallbladders secondary to giogram at the time of initial surgery were not available.
intentional or unintentional subtotal cholecystectomy along Each of the 12 patients had symptoms that persisted or
with patients truly having retained calculi in the cystic duct recurred after their initial cholecystectomy, similar in quality
,
stump.9 10 Estimating the actual incidence of remnant cystic to the pain experienced at their initial presentation. All patients
duct lithiasis is difficult, if not impossible, because patients reported right upper quadrant or epigastric pain. Four patients
may obtain follow-up care from centers distinct from where reported nausea/emesis. Three patients were discovered to
the original procedure was performed. have varying degrees of jaundice. Of note, 75 % of patients
Given the difficulty in determining the burden of disease had derangements in ≥1 indices on a standard liver enzyme
attributable to RCDL, we designed a study to evaluate our panel (total bilirubin, aspartate aminotransferase [AST], ala-
institutional experience with RCDL and herein describe the nine aminotransferase [ALT], alkaline phosphatase, and
largest case series to date specific for the condition. Our gamma-glutamyl transferase [GGT]). The most common lab-
primary goal was to define risk factors associated with oratory abnormality occurred in GGT (abnormal in 80 % of
RCDL. Secondarily, while the historical gold standard for those tested), followed by AST, ALT, and alkaline phospha-
the treatment of RCDL has been surgical excision of the cystic tase (each abnormal in 58.3 %) (Table 1).
duct remnant, we describe our experience in diagnosing and
treating these patients using a multidisciplinary approach. Table 1 Liver enzyme panels for each patient with remnant cystic duct
stump calculi at time of discovery

Patient TBili AST ALT AlkPhos GGT

Materials and Methods 1 0.3 25 22 10 20


2 4.1 96 316 415 527
We conducted a retrospective study of all patients un- 3 1.4 67 209 160 316
dergoing cholecystectomy or endoscopic retrograde 4 0.7 103 128 150 157
cholangiopancreatography (ERCP) from August 2001 to 5 1.3 92 370 70 N/A
May 2012 at the University of North Carolina (UNC) 6 1.5 156 269 181 311
Hospitals. Electronic medical record and endoscopy databases 7 3.0 925 819 185 889
were queried. Patients who underwent surgery for RCDL, or 8 0.1 17 23 73 N/A
patients identified at ERCP as having RCDL, were selected 9 1.3 31 49 125 190
for study inclusion in this case series. For each subject, we 10 0.7 32 44 130 159
recorded demographic information, existing comorbidities, 11 0.7 144 115 102 401
preoperative and postoperative symptoms, imaging results, 12 0.6 31 30 127 15
operative and pathologic findings, postoperative complica- Total abnormal (%) 50 58.3 58.3 58.3 80
tions, and mortality. Follow-up was conducted using postop-
erative clinic notes to monitor for recurrent symptoms. Elevated values are listed in bold
Statistical analysis was not performed given the small number TBili total bilirubin (upper limit of normal 1.2 mg/dL), AST aspartate
aminotransferase (upper limit of normal 55 U/L), ALT alanine amino-
of patients identified with the disease process. This study was transferase (upper limit of normal 72 U/L), AlkPhos alkaline phosphatase
approved by the Institutional Review Board of the UNC (upper limit of normal 126 U/L), GGT gamma-glutamyl transferase
School of Medicine. (upper limit of normal 68 U/L), N/A not available
J Gastrointest Surg

Initial operations consisted of nine laparoscopic and three identifiable. The relative utilization of the various common
open cholecystectomies. No choledocholithiasis or cystic duct imaging modalities is presented in Table 2.
calculi were noted in any of the 12 initial cholecystectomies. Management of RCDL included several modalities. There
Intraoperative cholangiography was performed in only one was an equal distribution in the number of patients undergoing
cholecystectomy (laparoscopic) without discovery of cystic reoperation (six) compared to endoscopic treatment alone
duct lithiasis. While the actual diameter of the cystic duct was (six). Two of the six patients represented with the clinical
available in only three operative reports (5, >10, >10 mm), constellation of symptoms and findings consistent with acute
eight operative reports made specific notation that the cystic remnant cystic duct obstruction and inflammation that re-
duct was noticeably dilated at the time of cholecystectomy. quired urgent surgical intervention. These symptoms and
Only one operative report stated that the cystic duct was findings included persistent right upper quadrant pain for
normal in caliber. >24 h, nausea and emesis, abnormal LFTs, leukocytosis, and
Cystic duct ligation techniques were recorded. Five cystic cystic duct cholelithiasis with inflammatory changes on radio-
ducts were ligated in what is considered “usual” laparoscopic graphic imaging. Due to the nature of clinical presentation, the
means at our institution (application of standard titanium clip patients were taken directly to surgery without attempted
or Hem-o-lok® clip [Teleflex, Research Triangle Park, NC]). endoscopic intervention. Of the remaining four patients
Of the remainder, four cystic ducts required a stapler transec- treated surgically, three had failed attempts to remove stones
tion, one required an Endoloop® (Ethicon Endo-Surgery, endoscopically, including one patient with Mirizzi syndrome
Cincinnati, OH), and two required hand-sewn closure tech- and one patient with extravasation of contrast at initial ERCP.
niques. Both the use of Endoloop® and suture ligation tech- The final patient elected to proceed to surgery without
niques were noted in the operative report to be necessary due attempted endoscopic stone retrieval. Of the patients
to the enlarged diameter of the cystic duct. undergoing repeat surgery, all were attempted laparoscopically
Two patients developed cystic duct stump leaks, also but required conversion to an open procedure due to
known as Strasberg type A bile duct injuries,11 after the initial dense adhesions/chronic inflammation in the porta hepatis.
surgery. Both injuries were successfully treated by percutane- Intraoperative cholangiography revealed the presence of con-
ous drainage. Radiologic imaging was utilized extensively in comitant choledocholithiasis in both patients, and a common
the evaluation of PCS in our series. Of all imaging modalities, bile duct exploration was employed for calculi removal at the
magnetic resonance cholangiopancreatography (MRCP) was time of reoperation. Six patients were managed by ERCP;
the most often utilized to obtain the diagnosis of RCDL (six). three with sphincterotomy and balloon extraction; and one
The remaining calculi were discovered by computerized to- with sphincterotomy, lithotripsy, and basket retrieval. One
mography (CT) scan (two), ultrasonography (two), operative patient required only balloon sweep of the common bile duct.
cholangiography (one), and ERCP (one) (Fig. 1). In most In the remaining patient, the cystic duct stone could not be
patients, there were multiple radiologic imaging modalities readily retrieved by ERCP and no further management was
utilized in the evaluation process, with no discernible order employed due to the patient’s preference.

Fig. 1 Retained cystic duct


calculi identified by endoscopic
retrograde cholangiography (a)
and MRCP (b). Arrow denotes
filling defects identified in each
imaging modality
J Gastrointest Surg

Table 2 Radiologic imaging modalities utilized in the evaluation of us to accurately state the incidence of symptomatic RCDL on
postcholecystectomy syndrome and their accuracy in identifying remnant
the basis of our single-institution study.
cystic duct lithiasis (RCDL)
In this series, symptoms associated with PCS in cases of
Radiologic Patients undergoing Number of Accuracy RCDL, specifically right upper quadrant pain, appear to be
study imaging modality RCDL identified (%) associated with dilation of a hollow viscus, as there was a high
Ultrasonography 9 5 55.6
rate of symptom resolution after both endoscopic and surgical
CT scan 7 5 71.4
interventions. Surgical pathology results and dense adhesions
noted at the time of surgery suggest that the presence of RCDL
MRI 9 8 88.9
leads to associated portal inflammation. However, the resolution
of PCS in 77.8 % of patients in this series, regardless of method
of treatment, suggests that ongoing symptoms are related to the
Of the 12 patients treated with surgical or endoscopic presence of stones themselves within the hollow viscus.
intervention for RCDL at our institution, 9 were seen in Proposed risk factors for recurrent symptoms due to RCDL
follow-up clinic visits by the attending gastroenterologist or include a cystic duct stump >1 cm in length, a low cystic duct
final operative surgeon. Two patients were lost to follow-up. insertion into the common bile duct, and use of nonabsorbable
, ,
One patient did not live local to our institution and after material for ligation of the cystic duct stump.9 13 14 The ac-
incomplete endoscopic treatment preferred no further endo- ceptable length of a cystic duct remnant in the laparoscopic era
scopic intervention or surgical referral. Of the nine patients has been an area of discussion due to the fact that one technical
with available follow-up clinic notes, seven experienced aspect of a safe dissection within the hepatocystic triangle is to
symptom resolution (77.8 %) with a mean follow-up of delineate the cystic duct at the cystic duct/gallbladder-infun-
11.8 months. Of the two patients with persistent symptoms, dibulum junction. In theory, this minimizes the chance for
notes suggested that one patient may have had pain from fatty injury of the common bile duct. However, if the cystic duct is
liver disease or a question of right upper quadrant incisional ligated in this location, there is often a long cystic duct
hernia and the other patient had ongoing work-up for persis- remnant present. Previous studies have concluded that in more
tent RCDL after initial endoscopic lithotripsy. than 50 % of cholecystectomies, the length of the remnant
cystic duct is ≥3 cm.15 With our current series, endoscopic and
surgical reports did not describe the exact location of the
Discussion stones in relation to the site of surgical transection or the
origin of the cystic duct. One patient was noted to have a large
The burden of disease attributable to RCDL as a cause of stone within the distal cystic duct remnant causing Mirizzi
postcholecystectomy syndrome has not been well-defined, syndrome at the time of representation. This makes it difficult
and estimates vary in the literature. It is difficult to quantify to describe the exact relationship between cystic duct length
the exact incidence at our institution, but RCDL is a rare cause and the occurrence of RCDL. In our opinion, given the po-
of postcholecystectomy syndrome in our patient population. tentially devastating consequences of common bile duct inju-
Rozsos et al. estimated that the cystic duct stump syndrome, or ry, a long cystic duct remnant confers less risk than aggressive
an excessively long cystic duct stump, is the cause of persis- dissection near the common bile duct. This assertion is
tent or recurrent symptoms in 16 % of patients with PCS.12 strengthened by the fact that several of the patients in our
There was no indication in this study regarding how many study with RCDL were managed by endoscopic therapies
patients had RCDL versus an isolated cystic duct stump alone.
without calculi. In a recent study by Palanivelu et al., the An interesting observation in our series was that the dictat-
incidence of remnant cystic duct stump calculi causing symp- ing surgeons noted a subjectively dilated cystic duct in 66.7 %
toms in patients undergoing standard laparoscopic cholecys- of these cases, with two estimated to be >1 cm in diameter.
tectomy was estimated at 0.02 %.9 However, this estimation This parallels the likelihood of surgeons utilizing special
was based on the number of patients undergoing remnant techniques for ligation of the cystic duct at the time of chole-
resection at their institution and did not include patients un- cystectomy, with 58.3 % choosing either a stapling device,
dergoing successful endoscopic retrieval. In addition, it is ligation loop, or suturing method over titanium or Hem-o-lok
unclear from this study how many patients had their initial clips, as is common at our institution. This detail may
operation at the same institution as the cystic duct stump be an important indicator to the surgeon about the
excision, and thus, the denominator used may not be repre- possibility of calculi within the cystic duct itself that should
sentative of the true extent of the disease burden. This exact prompt further diagnostic modalities, such as use of intraop-
situation is frequently encountered at our institution, and erative cholangiography.
nearly half of the patients in the present series had their initial Notably, only one patient undergoing cholecystectomy in
cholecystectomy at another facility. Thus, it is not possible for our study had a concomitant cholangiogram performed at the
J Gastrointest Surg

Fig. 2 Current institutional


algorithm used for the
management of
postcholecystectomy syndrome
and remnant cystic duct lithiasis

time of initial operation. Despite numerous well-designed studies speaks to the potential difficulty of this diagnosis and the need of
evaluating the value of intraoperative cholangiography, there is a high index of suspicion in patients with persistent symptoms.
still a lively discussion regarding the utility of routine versus
selective cholangiography. It seems reasonable to suggest that if a
selective approach is used for the identification of choledocholi- Conclusion
thiasis, then a dilated cystic duct may be a potential indication to
,
proceed with cholangiography. However, the utility and predic- Management of RCDL has traditionally been surgical.8 16
tive value of this strategy would need further investigation. With the advent of advanced minimally invasive techniques,
Radiologic imaging guidelines for the evaluation of pa- successful laparoscopic management of this condition has
,
tients with suspected PCS do not exist. Previous studies been reported,9 17 and our institution’s current algorithm re-
evaluating the diagnostic accuracy of various radiologic im- flects these changes (Fig. 2). In our experience, the six patients
aging modalities have found ultrasonography and MRCP to who required operative intervention were ultimately treated
be 60 and 92 % accurate, respectively.9 Our results are similar, through an open approach after conversion from laparoscopy.
showing the accuracy of ultrasonography and MRCP at 55.6 In all patients, the reason for conversion was dense chronic
and 88.9 %, respectively. While these findings support the use inflammatory tissue in the porta hepatis and inability to safely
of MRCP as the imaging modality of choice in the evaluation visualize the vasculo-biliary anatomy. Our institutional bias in
of suspected PCS, however, the additional cost of MRCP as these situations is for early conversion to open techniques to
well as the availability and local expertise regarding the inter- minimize the potential risk for vasculo-biliary injury, although
pretation of MCRP images must be considered. Anecdotally, we acknowledge that this is institution-dependent. Of note, six
our initial strategy for use of radiologic imaging began with of the patients in our series were managed by endoscopic
the least expensive and most readily available option, ultraso- techniques with successful calculi extraction from the cystic
nography. If this was unrevealing, then computerized tomog- duct remnant. This approach has previously been described,
raphy and magnetic resonance imaging were employed for but the actual utilization and availability of endoscopic tech-
further investigation. Although the accuracy of ultrasonogra- niques are not clearly defined.18 To our knowledge, this high
phy is lower than MRCP, it may be a more reasonable option rate of successful endoscopic management of remnant cystic
in the initial evaluation, when other diagnoses are being duct calculi has not been described before. In our experience,
entertained. Our current institutional algorithm omits the use endoscopy is a reasonable initial treatment option as it pro-
of ultrasonography in postcholecystectomy patients with ab- vides diagnostic and in some cases therapeutic alternatives.
normal liver function tests (Fig. 2). The delay of nearly 4 years The endoscopic approach would be favored as initial therapy
in our study between cholecystectomy and diagnosis of RCDL in patients presenting without recurrent inflammation of the
J Gastrointest Surg

remnant cystic duct. The only contraindications to endoscopic 4. Goenka MK, Kochhar R, Nagi B, Bhasin DK, et al. Endoscopic
retrograde cholangiopancreatography in postcholecystectomy syn-
intervention would be those associated with the endoscopic
drome. J Assoc Physicians India 1996;44:119-22.
procedure itself (inability to cannulate the sphincter of Oddi, 5. Rogy MA, Fugger R, Herbst F, Schulz F. Reoperation after chole-
coagulopathy, etc.). The early involvement of a biliary cystectomy. The role of the cystic duct stump. HPB Surg 1991;4:129-
endoscopist in the evaluation and treatment of PCS is crucial 34; discussion 34-5.
6. Stefanini P, Carboni M, Patrassi N, Loriga P, et al. Factors influencing
to the successful management of RCDL and may obviate the
the long term results of cholecystectomy. Surg Gynecol Obstet
need for repeat surgery altogether. 1974;139:734-8.
In conclusion, RCDL can be a cause of postcholecystectomy 7. Gui GP, Cheruvu CV, West N, Sivaniah K, et al. Is cholecystectomy
syndrome and the diagnosis should be entertained in a patient effective treatment for symptomatic gallstones? Clinical outcome
after long-term follow-up. Ann R Coll Surg Engl 1998;80:25-32.
with persistent or recurrent symptoms after cholecystectomy.
8. Walsh RM, Ponsky JL, Dumot J. Retained gallbladder/cystic duct
The ultimate diagnosis of RCDL is difficult and requires a remnant calculi as a cause of postcholecystectomy pain. Surg Endosc
multidisciplinary approach, for successful management. In 2002;16:981-4.
many circumstances, surgery can be avoided by endoscopic 9. Palanivelu C, Rangarajan M, Jategaonkar PA, Madankumar MV,
et al. Laparoscopic management of remnant cystic duct calculi: a
cystic duct stone retrieval methods.
retrospective study. Ann R Coll Surg Engl 2009;91:25-9.
10. Pernice LM, Andreoli F. Laparoscopic treatment of stone recurrence
in a gallbladder remnant: report of an additional case and literature
review. J Gastrointest Surg 2009;13:2084-91.
Conflict of Interest All authors declare that they have no conflicts of
11. Strasberg SM, Hertl M, Soper NJ. An analysis of the problem of
interest. This paper has not been published previously.
biliary injury during laparoscopic cholecystectomy. J Am Coll Surg
1995;180:101-25.
Funding Institutional support was provided by the Department of 12. Rozsos I, Magyarodi Z, Orban P. [Cystic duct syndrome and mini-
Surgery at the University of North Carolina at Chapel Hill. There are no mally invasive surgery]. Orv Hetil 1997;138:2397-401.
relevant financial disclosures. 13. Sitenko VM, Nechai AI, Stukalov VV, Kalashnikov SA. [Large
stump of the cystic duct]. Vestn Khir Im I I Grek 1976;116:56-9.
14. Freud M, Djaldetti M, De Vries A, Leffkowitz M. Postcholecystectomy
syndrome: a survey of 114 patients after biliary tract surgery.
References Gastroenterologia 1960;93:288-93.
15. Sezeur A, Akel K. Cystic duct remnant calculi after cholecystectomy.
J Visc Surg 2011;148:e287-90.
1. Russo MW, Wei JT, Thiny MT, Gangarosa LM, et al. Digestive and 16. Glenn F, McSherry CK. Secondary abdominal operations for symptoms
liver diseases statistics, 2004. Gastroenterology 2004;126:1448-53. following biliary tract surgery. Surg Gynecol Obstet 1965;121:979-88.
2. Zhou PH, Liu FL, Yao LQ, Qin XY. Endoscopic diagnosis and 17. Chowbey PK, Bandyopadhyay SK, Sharma A, Khullar R, et al.
treatment of post-cholecystectomy syndrome. Hepatobiliary Laparoscopic reintervention for residual gallstone disease. Surg
Pancreat Dis Int 2003;2:117-20. Laparosc Endosc Percutan Tech 2003;13:31-5.
3. Mergener K, Clavien PA, Branch MS, Baillie J. A stone in a grossly 18. Benninger J, Rabenstein T, Farnbacher M, Keppler J, et al.
dilated cystic duct stump: a rare cause of postcholecystectomy pain. Extracorporeal shockwave lithotripsy of gallstones in cystic duct
Am J Gastroenterol 1999;94:229-31. remnants and Mirizzi syndrome. Gastrointest Endosc 2004;60:454-9.

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