Sharp recanalization of CBD occlusion in patient with second orthotropic liver transplant with cocoon syndrome

Abstract

Biliary complications after orthotopic liver transplant (OLT) remain one of the primary causes of morbidity and mortality in liver transplant recipients with an approximate incidence between 5% and 32%. Given the limited supply of hepatic grafts, one of the most feared outcomes as a result of biliary complications is acute and or chronic graft failure. Biliary complications include leaks, biliary stasis, and stone formation, sphincter of Oddi dysfunction, recurrence of biliary disease (primary sclerosing cholangitis and primary biliary cirrhosis), and biliary strictures/obstruction. Overwhelmingly, the most common complication in hepatic transplantation is biliary stricture formation accounting for more than 50%. Currently, the mainstay of therapy as it pertains to biliary strictures/obstruction includes endoscopic retrograde cholangiography–guided therapy, percutaneous transhepatic cholangiography–guided therapy, or surgical revision/retransplantation. We present a case of biliary obstruction in a patient with a second liver transplant complicated by Cocoon Syndrome managed via sharp recanalization of CBD occlusion and placement of an endoscopic biliary Viabil stent.

Case presentation

We present a 66-year-old man with a history of alcoholic cirrhosis and hepatocellular carcinoma who presented with an incarcerated umbilical hernia necessitating surgical intervention. Postoperatively the patient experienced decompensated liver failure and orthotopic liver transplantation was performed with duct-duct biliary reconstruction. After liver transplantation, the patient developed hyperbilirubinemia, and an anastomotic stricture was identified on MRCP. An ERCP was performed and a stent was placed to alleviate the biliary stricture. A liver biopsy demonstrated bile ductular proliferation with periductal neutrophilic response with portal edema consistent with biliary outflow impairment. Unfortunately, after a protracted clinical course with resultant further hepatic decompensation and failure, repeat orthotopic liver transplantation was performed. A postoperative bowel obstruction was complicated by cocoon syndrome which required lysis of adhesions. The presumed etiology of the cocoon syndrome was recurrent peritonitis. Postop course was complicated by persistent hyperbilirubinemia. ERCP was unsuccessful. A percutaneous transhepatic cholangiography and a biliary drain placement were performed which showed a complete occlusion of the distal CBD and could not be crossed (Fig. 1). Sharp recanalization of the CBD was planned as the patient was not a candidate for third liver transplant.

Fig. 1.

Occlusion of the common bile duct (white arrow). Catheter in lesser. Sac draining bile (black arrow).

Technique

Existing 8F biliary drainage catheter was exchanged for an 8F sheath. 5F Berenstein catheter (Merit Medical, Salt Lake City, UT) loaded with 21 G Chiba needle (Cook Incorporated, Bloomington, IN) was advanced through the sheath to the point of occlusion (Fig. 1). Berenstein catheter was unsheathed while exposing the 21G Chiba needle. 10 F guiding catheter was advanced through the mouth, esophagus, and stomach into the duodenum. Gallstone retrieval basket was advanced through the sheath into the duodenum. A 21G Chiba needle was aimed at the basket after triangulating fluoroscopically (Fig. 2). Once the needle was captured by the basket, the stylet was removed and 300 cm long V18 wire (Boston Scientific, Marlborough, MA) was passed through the needle and snared by the basket (Fig. 3). The tract was dilated with a 10 mm by 4 cm high-pressure balloon and then stented with an endoscopic 10 mm by 4 cm Viabil stent (Gore Medical, Newark, DE) (Fig 4,5). The stent was then dilated with a 10 mm by 4 cm high-pressure balloon. A 10 F Internal external biliary drainage catheter (Uresil, Skokie, IL) was placed. Post-procedure Cholangiogram demonstrated contrast draining into the duodenum (Fig. 6).

Fig. 2.

Chiba needle (white arrow) aimed at the gallstone retrieval basket (black arrow).

Fig. 3.

V-18 wire (arrowhead) going through and through the sheath after being snared by the basket.

Fig. 4.

Track dilated by a 10 mm by 4 cm high-pressure balloon (black arrow).

Fig. 5.

A 10 mm by 4 cm Viabil stent placement (black arrow).

Fig. 6.

Cholangiogram through existing biliary drainage catheter shows contrast flowing through the Viabil biliary stent (white arrow) into the duodenum (black arrow).

Discussion

Biliary complications after orthoptic liver transplantation are common with incidence ranging between 5% and 32% [1]. In our case, the patient had a second orthoptic liver transplant complicated by cocoon syndrome and long-segment biliary obstruction. Cocoon syndrome is sclerosing encapsulating peritonitis where there is a total or partial encapsulation of abdominal viscera with fibrous membrane resulting in bowel obstruction. Our patient developed a small bowel obstruction postsecond OLT requiring extensive lysis of adhesions and was not a candidate for surgical reconstruction of biliary obstruction. ERCP was unsuccessful. The patient underwent a transhepatic biliary drainage, but the obstruction of the CBD could not be crossed. Subsequently, sharp recanalization of the common bile duct was planned and successfully performed. Sharp recanalization of central venous occlusion was first reported in 1999 by Farrell et al. It is a standard and acceptable procedure for central venous occlusion as per many studies including a retrospective cohort study that followed 37 patients each who underwent sharp recanalization for reestablishment of venous access. The procedure was found to have a success rate of 95% with sustainable positive outcomes [2]. Additional studies exist focused on various patient conditions that have been treated with sharp recanalization efficiently and safely. For example, a recent study of 27 sharp recanalization procedures performed for patients requiring hemodialysis with central venous occlusion had a success rate of 92.6% with limited complications [3]. There are cases reported in the literature where sharp recanalization was performed using radiofrequency wire and Chiba needle however, these were of short segment biliary occlusion [4], [5], [6]. Our patient similarly underwent a successful sharp recanalization of a long segment occlusion of the common bile duct to duodenum using a Viabil stent (Gore Medical). The patient unfortunately died from sepsis a few months later. A patent CBD stent with anastomosis to the duodenal bulb was seen at autopsy. Based on the success of the procedure in our patient and the success of other similar patients in the literature, sharp recanalization of a long-segment CBD occlusion should be considered as an option for an acceptable candidate.

Conclusion

To our knowledge, our case is the first to demonstrate the utility of sharp recanalization for the management of a long-segment biliary obstruction post OLT. Management of biliary complications post-OLT is essential to prevent liver failure. In our case, ERCP was unsuccessful and the patient was not a candidate for surgery due to cocoon syndrome. Sharp recanalization of a long-segment CBD occlusion is a safe alternative when endoscopic and surgical options are nonviable.

Patient consent

Patient expired due to sepsis. Informed consent was obtained from the patient's healthcare proxy for this publication