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| CASE REPORT |
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| Year : 2020 | Volume
: 10
| Issue : 1 | Page : 40-42 |
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Giant choledochal cyst presenting as cholecystocolonic fistula: A rarity
Md Mokarram Ali, Amit K Sinha, Amit Kumar, Rashi, Bindey Kumar
Department of Pediatric Surgery, AIIMS, Patna, Bihar, India
| Date of Submission | 27-Apr-2020 |
| Date of Acceptance | 06-Jun-2020 |
| Date of Web Publication | 06-May-2021 |
Correspondence Address:
Dr. Md Mokarram Ali
Department of Paediatric Surgery, AIIMS, Patna - 801 507, Bihar
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/ais.ais_23_20
Cholecystoenteric fistula is a rare entity, which is seen in cholecystitis complicated with cholangitis. The incidence of such fistula in pediatric patients with the choledochal cyst is extremely rare. Here, we report one such case of choledochal cyst in which cholecystoenteric fistula was detected intraoperatively.
Keywords: Cholangitis, cholecystocolonic fistula, choledochal cyst
How to cite this article:
Ali MM, Sinha AK, Kumar A, Rashi, Kumar B. Giant choledochal cyst presenting as cholecystocolonic fistula: A rarity. Arch Int Surg 2020;10:40-2 |
| Introduction | |  |
A choledochal cyst (CDC) is a rare congenital biliary tract anomaly characterized by cystic dilatation of the biliary tree. Patients with choledochal cyst who present after multiple episodes of cholangitis present with a challenge during surgery in the form of difficult dissection of the cyst. However, the incidence of cholecystoenteric fistula is rare in these patients. The association of this fistula with a choledochal cyst in the pediatric age group has not been reported yet. Here, we report a case of CDC in which cholecystocolonic fistula was an incidental finding during surgery.
| Case Report | | |
A 2-year-old female, presented with complains of abdominal distension, abdominal pain and recurrent vomiting for 6 months. There was no history of jaundice. On clinical examination, a soft cystic mass of size approximately 15 × 12 cm was palpable in right hypochondrium, which was extending up to the right iliac fossa. Ultrasonography revealed a large cystic lesion of size 9.1 × 7.4 cm at the site of porta hepatis. Further imaging was done in the form of contrast-enhanced computed tomography (CECT) of the abdomen, which was suggestive of large choledochal cyst type 1A [Figure 1]. Liver function tests were within normal range except for a slightly raised serum alkaline phosphatase (225.8). Total leukocyte counts were also raised with increased lymphocytes. | Figure 1: Contrast enhanced CT of abdomen showing type 1A choledochal cyst (arrow)
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After stabilization with antibiotics and intravenous fluid, the patient was taken for surgical excision of choledochal cyst with Roux-en-y hepaticojejunostomy. On exploration with right subcostal incision, type 1 choledochal cyst was found, which was densely adhered to the duodenum, transverse colon, and structures in the lesser sac. Gall bladder (GB) was found to be collapsed with a fistulous communication between the infundibulum of GB and transverse colon [Figure 2], [Figure 3]. The fistula was divided and the opening in colon repaired with absorbable suture. GB with choledochal cyst excised and Roux en Y hepaticojejunostomy was performed. Postoperative recovery was uneventful and the specimen was sent for histopathological examination. | Figure 2: Cholecystocolonic fistula (arrow) connecting infundibulum of gall bladder to transverse colon
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 | Figure 3: Cholecystoclonic fistula after division with visible gall bladder end (arrow) and colonic end
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| Discussion | | |
A biliary fistula is an abnormal communication of the biliary tract to any organ, cavity, or free surfaces. Fistula is classified as external (biliary-cutaneous) or internal (biliobiliary, bilioenteric, bronchobiliary).[1] Courvoisier described bilioenteric fistula in 1890. In adult literature, the incidence of these fistulas has been reported as 0.15%–-8%. The most common type of biliary enteric fistula is cholecysto-duodenal fistula (70%) followed by cholecystocolic fistula (8%–26%).[2],[3] However, the occurrence of these bilioenteric fistula and that too of a cholecystocolonic fistula in children has not yet been reported. There are very few case reports of cholecystocolonic fistula in adult patients with gallstones complicated by cholangitis.[4] These are often asymptomatic and often difficult to diagnose preoperatively. There is one case report of cholecystoduodenal fistula associated with a choledochal cyst in a 61-year-old female.[5] This fistula was detected intraoperatively, which was disconnected from the duodenum. The duodenal opening was closed and gall bladder along with choledochal cyst was excised.
Cholecystoenteric fistula as an associated congenital anomaly of the choledochal cyst has not been documented in literature so far. In our case, the cholecystocolonic fistula was incidentally detected intraoperatively, possibly formed due to multiple episodes of cholangitis, which were treated previously by oral medications. Computerized tomography of the abdomen, which was done for this patient, could not detect this fistulous tract.
The standard surgical procedure for type 1 and type 4 choledochal cyst is complete excision of cyst along with gall bladder followed by reconstruction of the biliary tree by Roux en Y hepaticojejunostomy. The abdomen can be approached via midline laparotomy incision or Chevron incision (preferred in children). After identifying the cyst, it is dissected all around to separate from the portal vein and hepatic artery. Dissection can proceed from a cephalic or caudal position. The gall bladder is dissected from the liver bed keeping intact with the common bile duct. Distally, the dissection is extended till intrapancreatic portion of duct, being careful not to injure the pancreatic duct. The distal-most portion of the cyst is ligated and divided proximal to it. The proximal end of the cyst is divided just distal to the confluence of the right and left hepatic duct. Bilioenteric continuity is achieved by Roux en Y hepaticojejunostomy, the jejunal loop being brought through the mesocolic window.
In the index case, we followed the standard surgical technique as described above. One extra step was the division of the chloecystocolonic fistula, which was identified incidentally. The colonic end was closed with interrupted absorbable sutures. The fistulous tract was removed along with gall bladder and cyst.
| Conclusion | | |
Cholecystocolonic fistula in association with the choledochal cyst is extremely rare in the pediatric age group. Even preoperative contrast-enhanced CT of abdomen fails to detect this lesion. It may present as surgical surprise and surgeons should be cautious, especially when dealing with choledochal cyst after multiple episodes of cholangitis.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the legal guardian has given his consent for images and other clinical information to be reported in the journal. The guardian understands that names and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
| 1. | Bhat GA, Jain R, Lal P. Cholecystoduodenocolic fistula: An unexpected intraoperative finding, a surgical challenge. Int J Clin Med 2016;7:261-4.  |
| 2. | Balent E, Plackett TP, Lin-Hurtubise K. Cholecystocolonic fistula. Hawai'i J Med Public Health 2012;71:155-7. |
| 3. | Angrisani L, Corcione F, Tartaglia A, Tricarico A, Rendano F, Vincenti R, et al. Cholecystoenteric fistula (CF) is not a contraindication for laparoscopic surgery. Surg Endosc 2001;15:1038-41. |
| 4. | Antonacci N, Taffurelli G, Casadei R, Ricci C, Monari F, Minni F. Asymptomatic cholecystocolonic fistula: A diagnostic and therapeutic dilemma. Case Rep Surg 2013;2013:754354. doi: 10.1155/2013/754354. Epub 2013 Apr 17. PMID: 23691423; PMCID: PMC3652046. |
| 5. | Banerjee Jesudason SR, Raju RS. Cholecystoduodenal fistula associated with choledochal cyst: A rare clinical entity. J Hepatobiliary Pancreat Surg 2008;15:664-6. |
[Figure 1], [Figure 2], [Figure 3]
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