Ruptured choledochal cyst: One-stage or two-stage, open or laparoscopic surgery? : Journal of Minimal Access Surgery

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Unusual Case

Ruptured choledochal cyst: One-stage or two-stage, open or laparoscopic surgery?

Ojha, Sunita1,; Agarwala, Pooja2; Sharma, Ravi2; Bharadia, Lalit2

Author Information
Journal of Minimal Access Surgery 19(1):p 138-140, Jan–Mar 2023. | DOI: 10.4103/jmas.jmas_206_21
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Abstract

INTRODUCTION

Choledochal cyst (CDC) is a congenital anomaly of the biliary system requiring surgical excision and biliary reconstruction to avoid complications such as perforation, pancreatitis and malignancy. The first successful laparoscopic CDC excision and biliary reconstruction were performed in 1995.[1] Since then laparoscopy has been widely used due to its well-known advantages compared to open techniques for CDC.[2] Rupture/perforation of CDC is described in 2% of cases. In the case of ruptured CDC, there are different approaches regarding the stage of surgery (one stage/two staged). Laparoscopy is a procedure of technical challenge in ruptured CDC. We discuss here such a rare case managed with single-stage laparoscopic procedure.

CASE REPORT

An 18-month-old-female child weighing 8 kg, presented with fever, abdominal distension and respiratory discomfort for 5 days. She had feeble pulse, 160/min heart rate, tachypnoea, nasal flaring and severe abdominal distension. Leucocyte counts, bilirubin, gamma GT and alkaline phosphatase were raised. Ultrasonography showed thick-walled dilated common bile duct (CBD) (19 mm) with massive ascites. Computed tomography (CT) scan showed proximal dilated CBD, mild intrahepatic biliary radicals (IHBR) dilatation with ascites. The percutaneous abdominal drain was placed which drained 2 l of bile. General condition improved after drain placement, fluid resuscitation and antibiotics. She had 1–1.5 l bile drain/day. Endoscopic retrograde cholangiopancreatography (ERCP) stenting was not feasible in small child. Subsequently, child underwent laparoscopic CDC resection and hepatico-duodenostomy on day 4.

Five ports were placed. Umbilical (5 mm), right subcostal midclavicular (3 mm), anterior axillary line (3 mm), below xiphisternum (5 mm) and one slightly towards left of midline above the umbilical port (5 mm). Pneumoperitoneum was created using 8 mm-Hg pressure at flow of 8 l. Bowel was covered with greenish layer of bile [Figure 1a]. Adhesiolysis was done at the sub-hepatic area. Biliary sludge and calculi helped in identifying ruptured CDC (type 1B) [Figure 1]. The anterior wall of CDC was found ruptured with sloughed-out necrotic edges [Figure 2]. Oedematous and thickened cyst wall were dissected from medial to lateral aspect, making it free from portal vein [Figure 2]. Dissection was done close to the cyst wall to avoid vascular complications. The right hepatic artery was identified posterior to cyst. Distal thin end of the cyst was flushed with saline, clipped and transacted. Proximal end was transacted at the hepatic confluence. Hepatico-duodenostomy was done using interrupted PDS4-0 sutures starting from the lateral angle. Interrupted sutures avoid cut through of sutures especially in oedematous tissues, and causes less traction on the suture line while suturing. The gall bladder was used as traction to retract the liver till suturing was completed. Subsequently, cholecystectomy was done and the specimen was retrieved. The drain was placed posterior to the anastomosis. The duration of surgery was 190 min.

F1
Figure 1:
(a) layer of bile and biliary sludge over bowel and gallbladder. (b) Calculi and sludge seen at the ruptured anterior wall of choledochal cyst (at suction tip). (c) Ruptured choledochal cyst with visualised hepatic duct orifice, necrosed edges of choledochal cyst (arrow)
F2
Figure 2:
Sloughed out anterior wall of choledochal cyst with necrotic margins (black arrow), posterior wall of choledochal cyst is dissected free from portal vein (yellow arrow). Distal narrow end (white arrow) and proximally hepatic duct orifice at the confluence is seen. GB: Gallbladder

Oral feeds were started on the 3rd postoperative day (POD). Drain removed on 4th POD and discharged on the 6th POD. In the follow-up of 1 year, the child is asymptomatic.

DISCUSSION

Biliary peritonitis secondary to rupture of CDC is a rare complication. Increased intraluminal pressure due to protein plug, or destructive effect of refluxing pancreatic juices on bile duct, is considered to cause weakening in the wall of the bile duct, leading to perforation.[3] Bile on paracentesis and sonography/CT/magnetic resonance imaging showing dilated common bile-duct with IHBR suggests perforated CDC. Various methods of management have been proposed as surgical treatment is difficult and has risk of complications.[4] Biliary drainage by T-tube placement, cholecystostomy/choledochostomy, lavage followed by definitive surgery after 4–6 weeks are proposed two-stage treatments.[3]

A single-centre experience with 27 cases of perforated CDC advocated single-stage surgery, avoiding discomfort and morbidity of weeks of carrying biliary drainage/T-tube. Challenges of single-stage surgery are technical difficulty in dissection, total excision of CDC leading to the risk of anastomotic leak. Potential hesitancy in performing single-stage surgery is not justified, as the comparison between one-stage and two-stage surgery showed statistically no significant difference.[5]

Although one-stage surgery or laparoscopy is not indicated in the sick unstable patient,[4] our patient would still have required surgery for biliary drainage and peritoneal lavage. Hence, we used percutaneous peritoneal drainage as bridging procedure to stabilise the patient. ERCP was not feasible in small child and would not have been of much help as the anterior wall of CDC was necrotic and sloughed out. Adhesions over 6-week period could have further increased technical difficulty in both laparoscopy or laparotomy. Adequate training in laparoscopic surgery, proper delineation of anatomy, judicious use of electrocautery, will help in avoiding mishaps.

To the best of our knowledge, this is the first case report of single-stage laparoscopic CDC excision and reconstruction for ruptured CDC.

CONCLUSION

Single-stage laparoscopic surgery in ruptured CDC is technically demanding, but good outcome is feasible with proper planning and expertise.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient (s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initial s will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

Acknowledgement

We acknowledge the contribution of Dr. Rajiv Bansal and Dr. Anupam Chaturvedi for technical and material support.

REFERENCES

1. Farello GA, Cerofolini A, Rebonato M, Bergamaschi G, Ferrari C, Chiappetta A Congenital choledochal cyst:Video-guided laparoscopic treatment. Surg Laparosc Endosc 1995;5:354–8.
2. Sun R, Zhao N, Zhao K, Su Z, Zhang Y, Diao M, et al. Comparison of efficacy and safety of laparoscopic excision and open operation in children with choledochal cysts:A systematic review and update meta-analysis. PLoS One 2020;15:e0239857.
3. Ando K, Miyano T, Kohno S, Takamizawa S, Lane G Spontaneous perforation of choledochal cyst:A study of 13 cases. Eur J Pediatr Surg 1998;8:23–5.
4. Chiang L, Chui CH, Low Y, Jacobsen AS Perforation:A rare complication of choledochal cysts in children. Pediatr Surg Int 2011;27:823–7.
5. Ngoc Son T, Thanh Liem N, Manh Hoan V One-staged or two-staged surgery for perforated choledochal cyst with bile peritonitis in children?A single center experience with 27 cases. Pediatr Surg Int 2014;30:287–90.
Keywords:

Complicated choledochal cyst; laparoscopy in ruptured choledochal cyst; one-stage surgery for ruptured choledochal cyst; ruptured/perforated choledochal cyst

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