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CASE REPORT
Year : 2020  |  Volume : 8  |  Issue : 3  |  Page : 152-155

Cystocholecystostomy: A new technique for management of hilar biliary disruption due to a large hydatid cyst liver - Ten years of follow-up


Department of General Surgery, Pt. B. D. Sharma Post Graduate Institute of Medical Sciences, Rohtak, Haryana, India

Date of Submission27-May-2021
Date of Acceptance22-Jun-2021
Date of Web Publication19-Jul-2021

Correspondence Address:
Dr. Sanjay Marwah
Department of General Surgery, Pt. B. D. Sharma Post Graduate Institute of Medical Sciences, Rohtak - 124 001, Haryana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ssj.ssj_73_21

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  Abstract 

A long-standing and large-sized hepatic hydatid cyst located at the liver hilum is likely to rupture into the adjoining major bile ducts. This unusual complication demands preoperative diagnosis at an early stage and adequate surgical management so as to prevent morbidity and mortality. Such cases are managed with bilioenteric anastomosis by doing Roux-en-Y cystojejunostomy. However, one such case was managed by performing side to side anastomosis between disrupted hydatid cyst at the hilum and adjoining gallbladder. This innovative surgical procedure was named “Cysto-cholecystostomy” and was reported for the first time in the English literature. This case report presents ten years of follow up of this patient along with discussion of related cases published in the literature.

Keywords: Cystocholecystostomy, hepatic hydatid cyst, intrabiliary rupture


How to cite this article:
Marwah S, Singh R, Mandal A, Das S. Cystocholecystostomy: A new technique for management of hilar biliary disruption due to a large hydatid cyst liver - Ten years of follow-up. Saudi Surg J 2020;8:152-5

How to cite this URL:
Marwah S, Singh R, Mandal A, Das S. Cystocholecystostomy: A new technique for management of hilar biliary disruption due to a large hydatid cyst liver - Ten years of follow-up. Saudi Surg J [serial online] 2020 [cited 2021 Dec 8];8:152-5. Available from: https://www.saudisurgj.org/text.asp?2020/8/3/152/321737


  Introduction Top


Hydatid disease most commonly involves the liver in 50%–70% of the cases.[1] The disease remains asymptomatic in majority of the cases. The symptoms usually appear due to the occurrence of complications such as suppuration and intrabiliary rupture of the cyst.[2] Such cases need surgical removal of the cyst contents with entero-anastomoses such as cystojejunostomy or cystogastrostomy depending on the position of the cyst.[3] We reported one such case that was successfully managed with a new and simple operative technique of cystocholecystostomy.[4] After 10 years, there are no complications related to this previously unreported anastomotic technique in this patient.


  Case Report Top


A 35-year-old male with a hydatid cyst liver was admitted in emergency in a state of shock with obstructive jaundice. He improved after resuscitation and on investigations, a large hydatid cyst measuring 8 cm × 8 cm in the right lobe of the liver near the hilum was found that had ruptured into the biliary system [Figure 1] and [Figure 2]e. After 1 month of oral albendazole with proper informed and written consent he underwent exploratory laparotomy with a plan to perform Roux-en-Y hepaticojejunostomy. The operative findings revealed 10 cm × 8 cm, thick-walled hydatid cyst in the right lobe of the liver near the hilum. The cyst was deroofed, contents were evacuated, and the cavity was washed with normal saline that revealed stretched and divided ends of the right hepatic duct draining clear bile.
Figure 1: Preoperative MRCP showing ruptured hydatid cyst into the right hepatic duct. The right hepatic duct and its branches are displaced and dilated, whereas the left hepatic duct is normal[4]

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Figure 2: (a) End of T-tube (arrow) seen projecting into de-roofed cyst cavity through the common bile duct. GB – Gallbladder.[4] (b) Gallbladder mobilized and opened longitudinally (arrow) against cyst cavity.[4] (c) Gallbladder sutured with the cyst wall using 2-0 interrupted vicryl suture (arrow).[4] (d) Cystocholecystostomy completed (white arrow) with T-tube in the common bile duct (black arrow).[4] (e) Line diagram showing ruptured hydatid cyst into the right hepatic duct.[4] (f) Line diagram showing mobilized gallbladder with cystocholecystostomy and T-tube in situ[4]

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However, once the cyst wall was opened, there was a spillage of its contents into the peritoneal cavity leading to anaphylactic shock. Supraduodenal choledochotomy was done and Kehr's T-tube was placed with its proximal limb reaching into the cyst cavity through the ruptured right hepatic duct [Figure 2]a. The gallbladder of the patient was normal looking and its fundus was very close to the opened up cyst cavity. After dissection, the gallbladder was made to overlap the cyst cavity and opened with a longitudinal incision from the fundus to Hartmann's pouch [Figure 2]b. A tension-free side-to-side anastomosis was performed between hydatid cyst and gallbladder using interrupted 2-0 vicryl sutures [Figure 2]c, [Figure 2]d and [Figure 2]f. This surgical procedure was named “cysto-cholecystostomy” and was performed for the first time ever as an emergency life-saving procedure. The postoperative period was largely uneventful. The T-tube was removed at 6 weeks after confirming the patency of cystocholecystostomy anastomosis on magnetic resonance cholangiogram [Figure 3]a. The patient was given oral albendazole for 6 months. At 1½ years after surgery, Magnetic resonance cholangiopancreatography (MRCP) was done that revealed an almost resolved cyst cavity draining well into the gallbladder through cystocholecystostomy with no dilatation of intrahepatic biliary radicles [Figure 3]b. Thereafter, the patient was lost to follow-up.
Figure 3: (a) MRCP at 6 weeks after surgery demonstrating patent cystocholecystostomy anastomosis (white arrow) with shrunken cyst (red arrow) and T-tube in situ.[4] (b) MRCP at 1.5 years showing patent cystocholecystostomy anastomosis (arrow). The cyst cavity has completely resolved[4]

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The patient presented again in emergency 10 years later with acute alcoholic pancreatitis [Figure 4] and managed conservatively. On MRCP, the cystocholecystostomy was functioning well without any back pressure changes [Figure 5].
Figure 4: Contrast-enhanced computed tomography of the abdomen showing edematous pancreatitis

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Figure 5: MRCP film showing patent anastomosis between the gallbladder and right hepatic duct (red arrow). The gallbladder is marked with a yellow arrow. The hepatic hydatid cyst has shrunken and disappeared

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  Discussion Top


The hydatid cyst rupture into the biliary system is reported to occur in 5%–25% of the cases.[5] These cases usually present with features of acute cholangitis. The preoperative diagnosis is made on ultrasound, contrast-enhanced computed tomography, and MRCP. The management objective is to clear the contents of the cyst, manage the residual cyst cavity, clear debris from the biliary system, and restore free flow of bile.[6],[7],[8] The surgical options vary from radical procedures such as selective hepatic lobectomy and total/partial cystectomy followed by Roux-en-Y pericystojejunostomy to conservative procedures such as suture of cystobiliary fistula, double side drainage, and cystobiliary disconnection.[6] The hydatid debris present in the biliary system is managed with common bile duct exploration, irrigation and T-tube drainage, sphincteroplasty, or choledochoduodenostomy.[6],[7],[8] The most common postoperative complication is persistent external biliary fistula, best managed with endoscopic sphincterotomy.[5]

There are high chances of external biliary fistula when a large hilar hydatid cyst ruptures into major bile ducts. Such cases are best managed by creating internal biliary drainage with Roux-en-Y cystojejunostomy.[9] In the present case, on opening the cyst wall, accidental intraperitoneal spillage led to anaphylactic shock. Incidentally, the gallbladder was hanging loose near the opened up cyst cavity. It was decided intraoperatively to perform side-to-side anastomosis between fundus and ruptured right hepatic duct instead of Roux en Y hepaticojejunostomy to save time. The procedure of “cysto-cholecystostomy” was quick and the patient had uneventful recovery. This patient had no complication related to cystocholecystostomy up to 10 years of follow-up indicating acceptable long-term outcome.

In a recently published article, the authors have presented a series of twenty cases, over a period of 7 years, undergoing laparoscopic cystocholecystostomy for hepatic cysts with biliary communications and cystic dilatations of main intrahepatic ducts in a pediatric population.[10] No patient developed bile leak, anastomotic stenosis, stone formation, or cholangitis. Liver functions normalized postoperatively. The study concluded that laparoscopic cystocholecystostomy is a simpler and more physiological surgical alternative for managing such cases. This was a retrospective study giving good results without complications with up to 3 years follow-up. Unlike this study having planned cystocholecystostomy, the technique in our case was performed by default as a life-saving measure in a compromised patient and the anastomosis has performed well over a period of 10 years.

The definite advantages of this procedure are preservation of sphincter of Oddi function, avoidance of multiple GI anastomoses and their complications, and shorter operative time. However, it has limited application since it can only be used in and around the hepatic hilum where mobilized gallbladder can reach easily. The procedure is rare since there is only one series of twenty cases after our case report. However, a large-scale, prospective, comparative study is warranted to confirm the results of this procedure. A long-term follow-up is required to verify whether the gallbladder drains as well as Roux-Y cystojejunostomy does. The hepatobiliary surgeons must be made aware of this technique for application whenever indicated to establish it as a standard operative procedure.

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 Top

1.
Dumas R, Le Gall P, Hastier P, Buckley MJ, Conio M, Delmont JP. The role of endoscopic retrograde cholangiopancreatography in the management of hepatic hydatid disease. Endoscopy 1999;31:242-7.  Back to cited text no. 1
    
2.
Berbece S, Cristian Blajut C, Ciurea S, Tomulescu V, Popescu I. Surgical treatment of complicated liver echinococcosis – Our experience with 184 cases in 10 years. J Transl Med Res 2016;21:42-6.  Back to cited text no. 2
    
3.
Barros JL. Hydatid disease of the liver. Am J Surg 1978;135:597-600.  Back to cited text no. 3
    
4.
Marwah S, Pandey S, Shivran K, Singla P, Sharma H, Rohilla S. Cystocholecystostomy: A new technique for management of hilar biliary disruption due to a large hydatid cyst liver. OA Case Rep 2014;3:61.  Back to cited text no. 4
    
5.
Akaydin M, Erozgen F, Ersoy YE, Birol S, Kaplan R. Treatment of hepatic hydatid disease complications using endoscopic retrograde cholangiopancreatography procedures. Can J Surg 2012;55:244-8.  Back to cited text no. 5
    
6.
Lygidakis NJ. Diagnosis and treatment of intrabiliary rupture of hydatid cyst of the liver. Arch Surg 1983;118:1186-9.  Back to cited text no. 6
    
7.
Kornaros SE, Aboul-Nour TA. Frank intrabiliary rupture of hydatid hepatic cyst: Diagnosis and treatment. J Am Coll Surg 1996;183:466-70.  Back to cited text no. 7
    
8.
Ulualp KM, Aydemir I, Senturk H, Eyuboğlu E, Cebeci H, Unal G, et al. Management of intrabiliary rupture of hydatid cyst of the liver. World J Surg 1995;19:720-4.  Back to cited text no. 8
    
9.
Yildirgan MI, Başoğlu M, Atamanalp SS, Aydinli B, Balik AA, Celebi F, et al. Intrabiliary rupture in liver hydatid cysts: Results of 20 years' experience. Acta Chir Belg 2003;103:621-5.  Back to cited text no. 9
    
10.
Diao M, Li L, Cheng W. Cysto-cholecystostomy: A more physiological procedure for hepatic cysts with biliary communications and cystic dilatations of main intrahepatic ducts. World J Surg 2018;42:2599-605.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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