Title: Longterm outcome of rendezvous technique for hepaticojejunal anastomotic obstruction after pediatric living donor liver transplantation
Abstract: [The copyright line for this article was changed on March 13, 2020 after original online publication.] Hironori Yamamoto consults for and received grants from Fujifilm Corp. Yukihiro Sanada is responsible for the study design. Yukihiro Sanada, Tomonori Yano, Taizen Urahashi, Yoshiyuki Ihara, Noriki Okada, Naoya Yamada, Yuta Hirata, and Takumi Katano are responsible for acquisition of data. Yukihiro Sanada and Tomonori Yano are responsible for analysis and interpretation. Tomonori Yano, Hironori Yamamoto, and Koichi Mizuta are responsible for the revision of the text. TO THE EDITOR: Hepaticojejunal anastomotic obstruction (HJO) is diagnosed when contrast medium delivered via percutaneous transhepatic cholangiography (PTC) does not flow into the jejunum or when the hepaticojejunal anastomotic site cannot be identified using enteroscopy. HJO after liver transplantation (LT) is a rare biliary complication,1 and intractable HJO can lead to severe complications, including graft failure. Although surgical revision is the first choice for the treatment of HJO, it is an invasive procedure that can cause additional injury. With the advances in and benefits of endoscopic instruments and techniques, however, endoscopic treatments for hepaticojejunal anastomotic stricture (HJS) offer a promising less‐invasive procedure.3 Since the development of instruments and techniques for double‐balloon enteroscopy (DBE),4 it has been possible to perform endoscopic retrograde cholangiography despite the length of the necessary passage, the strong adhesion of the Roux‐en‐Y limb to the peritoneum, and the difficult angulation of the hepaticojejunal anastomosis. The penetration and balloon dilatation of the HJO using combined percutaneous transhepatic cholangioscopy (PTCS) and DBE is performed.2 The anastomotic penetration procedure for HJO combined with PTCS and DBE compose the so‐called “rendezvous technique.” Our institution has reported a pediatric case study of the rendezvous technique for HJO after living donor liver transplantation (LDLT).2 However, the longterm patency of the hepaticojejunal anastomosis after the rendezvous technique is still unclear. Therefore, in this study, we described our experience with the rendezvous technique for HJO after pediatric LDLT and the longterm outcomes after HJO treatment. Patients and Methods This study included 370 outpatients who underwent LT between October 1988 and December 2016 at Jichi Medical University and other facilities. Between October 2005 and February 2009, 9 endoscopic interventions using the rendezvous technique were performed for 6 (1.6%) patients with HJO after pediatric LDLT. We evaluated 6 patients (3 males and 3 females) with a median age of 12.5 years (range, 5.4‐17.2 years), and a median body weight (BW) of 33.3 kg (range, 15.1‐47.5 kg). The original disease was biliary atresia (BA) in all 6 patients. The posttreatment observation period ranged between 7.5 and 10.9 years. Approval to conduct this study was obtained from the ethics committees of Jichi Medical University (ethics committee approval case number 15‐106). DIAGNOSIS OF HJO HJO is diagnosed when the contrast medium delivered via PTC does not flow into the Roux‐en‐Y limb,5 when no real‐time moving images are obtained under fluoroscopy, or when the hepaticojejunal anastomotic site cannot be confirmed with DBE. THERAPEUTIC STRATEGY FOR HJO We present the therapeutic strategy for HJS or HJO in Fig. 1. The indication for DBE is more than 15 kg of BW because of instrumental and technical limitations. Therefore, observation and oral administration of ursodeoxycholic acid is the recommended strategy for patients who weigh <15 kg and for those with no dilatation of the intrahepatic bile duct (IHBD).Figure 1: Therapeutic strategy for HJS/HJO after LT.When HJO is diagnosed, treatment using the rendezvous technique with DBE and PTCS is performed after the percutaneous transhepatic biliary drainage (PTBD). In patients with HJO, if the treatment by the rendezvous technique is unsuccessful, surgical revision is performed. Results A total of 9 endoscopic interventions using the rendezvous technique were performed for 6 patients with HJO after pediatric LDLT (Table 1). The median period between the HJO treatment and the LDLT was 3.1 years (range, 0.7‐13.0 years). The rendezvous techniques employed were PTCS plus DBE in 5 patients and PTCS plus jejunography using DBE in 1 patient. Table 1 - Clinical Findings of the Patients Who Underwent the Rendezvous Technique for HJO After LDLT Case and Year Original Disease/BW (kg) Age at HJO Treatment (year) Period Between HJO Treatment and LDLT (year) Rendezvous Technique Outcome Prognosis and the Second Treatment Case 1 2005 BA/37.5 13.7 0.7 PTCS plus jejunography using DBE (EN‐450P5/20) Success Recurrence‐free for 10.9 years (10.5 years after removal of tube) Case 2 2006 BA/27.3 12.2 0.8 PTCS plus DBE (EC‐450BI5) Success Recurrence at 7 months 2007 BA/33.3 12.8 1.3 PTCS plus DBE (EC‐450BI5) Success Recurrence at 7 months 2008 BA/35.0 13.3 1.9 PTCS plus DBE (EC‐450BI5) Success Recurrence‐free for 8.6 years (7.9 years after removal of tube) Case 3 2007 BA/47.5 17.2 13.0 PTCS plus DBE (EN‐450P5/20) Success Recurrence‐free for 9.4 years (7.2 years after removal of tube) Case 4 2007 BA/38.7 12.5 7.6 PTCS plus DBE (EN‐450P5/20) Failure Surgical revision Case 5 2008 BA/18.9 6.9 3.1 PTCS plus DBE (EN‐450P5/20) Success Recurrence‐free for 7.7 years (7.2 years after removal of tube) Case 6 2009 BA/15.1 5.4 4.2 PTCS plus DBE (EN‐450P5/20) Failure Re‐rendezvous penetration method 2009 BA/15.1 5.5 4.3 PTCS plus DBE (EC‐450BI5) Success Recurrence‐free for 7.5 years (5.9 years after removal of tube) The initial successful intervention and recurrence‐free rates were 66.7% and 71.4%, respectively. The final successful intervention and recurrence‐free rates were 83.3% and 83.3%, respectively. The median recurrence‐free period was 8.7 years (range, 7.5‐10.9 years). Case 4, the patient in whom the hepaticojejunal anastomotic site could not be identified, underwent a surgical revision. Two episodes of HJO recurrence after the rendezvous technique occurred in case 2, and the patient underwent endoscopic interventions using the rendezvous technique 3 times. These HJO recurrences occurred after the PTBD tube was pulled out of the IHBD. During the first treatment, a guidewire presented via the PTC route penetrated the intra‐abdominal cavity on the first attempt; during the second attempt, a guidewire presented via the PTC route penetrated to the Roux‐en‐Y limb, and the hepaticojejunal anastomotic site was dilated. The PTBD tube was placed beyond the hepaticojejunal anastomotic site. The first recurrence of HJO occurred 5 months after the PTBD tube was placed. During the second treatment, a guidewire presented via the PTC route penetrated to the Roux‐en‐Y limb directly, and the hepaticojejunal anastomotic site was dilated on the first attempt. The second episode of HJO recurrence took place 5 months after the first recurrence. During the third treatment, a guidewire via the PTC route penetrated to the Roux‐en‐Y limb directly, and the hepaticojejunal anastomotic site was dilated on the first attempt. The PTBD tube was placed for 8 months and removed, and no further HJO recurrence was reported. Five patients who underwent the rendezvous technique were able to have their PTBD tube removed. The median PTBD tube‐free period was 7.5 years (range, 5.9‐10.5 years). The graft and patient survival rates were both 100%. Discussion The “rendezvous technique” has been reported as the anastomotic penetration procedure used for severe duct‐to‐duct biliary anastomotic stricture using combined PTCS and endoscopic retrograde cholangiopancreatography, but there are few case reports of HJO using combined PTCS and DBE.2 It took a median of 3.1 years after LDLT to treat HJO in the present study. The HJS might have occurred at an early stage after LDLT. However, the mild liver dysfunction or mild dilatation of IHBD is observed for a longterm period. Therefore, HJS potentially progresses to HJO, and the treatment for HJO may be intractable and difficult. In the present study, the final successful and recurrence‐free rates when the rendezvous technique is used to treat HJO after LDLT were 83.3% and 83.3%, respectively, and the median recurrence‐free period was 8.7 years (range, 7.5‐10.9 years). When the rendezvous technique was unsuccessful in the present study (cases 4 and 6), it was because we could not reach the hepaticojejunal anastomotic site using DBE. However, if 3‐dimensional identification of the hepaticojejunal anastomotic site using real‐time moving images obtained with combined PTC and jejunography had been possible for case 1, the rendezvous technique could have been safely performed. In case 4, we could not identify the hepaticojejunal anastomotic site by jejunography. In case 6, we could not reach the hepaticojejunal anastomotic site using DBE (EN‐450P5/20), and therefore, we changed the scope of DBE to EC‐450BI5 and succeeded. The recurrence of HJO after the rendezvous technique is an important problem, but its cause is unclear. Two episodes of HJO recurrence occurred after the PTBD tube was pulled out of the IHBD in case 2. We hypothesized that the cause of recurrence in the case 2 patient was mispenetration1 and the short‐term placement of a PTBD tube.2 During the first treatment, the contrast agent was administered to the Roux‐en‐Y limb under direct vision with DBE to image the precise anastomosis site using fluoroscopy. To avoid mispenetration when using the rendezvous technique, a clip should be placed adjacent to the anastomosis site. Regarding the short‐term placement of a PTBD tube, the incidence rate of recurrent HJS after PTBD treatment for posttransplant HJS was 11.1%. We performed 279 LDLTs between May 2001 and December 2016 and performed PTBD treatment for 18 patients with HJS. Only 2 patients out of 18 (11.1%) patients developed recurrent HJS after PTBD tube removal, and the median duration of PTBD tube placement was 8 (4‐25) months. Therefore, we think that the cause of the second recurrence in the case 2 patient was the short‐term placement of the PTBD tube. In addition, the PTBD tube was placed for 8 months after the third treatment; after it was removed, there was no HJS recurrence. In conclusion, it is important for the patients with HJO to penetrate the anastomosis site under direct vision and three‐dimensional identification using combined PTCS and DBE. The rendezvous technique for HJO after LDLT provides a safe and less‐invasive treatment than surgical revision and offers a high success rate and longterm patency of the hepaticojejunal anastomosis.