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Successful Treatment of Isolated Draining Intrahepatic Biliary Ducts in a Pediatric Liver Transplant Recipient Using Alcohol Sclerotherapy

2008; Elsevier BV; Volume: 19; Issue: 1 Linguagem: Inglês

10.1016/j.jvir.2007.08.033

1535-7732

Darice Liu, Antoinette S. Gomes, Sue V. McDiarmid,

Organ Donation and Transplantation

Alcohol sclerotherapy is useful for the treatment of biliary cutaneous fistulas in patients who have undergone hepatic resection. The same principle can be applied for transplant recipients with isolated draining biliary ducts. Percutaneous therapy of isolated draining biliary ducts with absolute alcohol (ie, 100% ethanol) was successfully performed in a 7-year-old patient who had undergone orthotopic liver transplantation. The increasing use of segmental living and split cadaveric liver grafts, with the subsequent increase risk in biliary complications, necessitates more efficient therapy for isolated draining bile ducts. Alcohol sclerotherapy is useful for the treatment of biliary cutaneous fistulas in patients who have undergone hepatic resection. The same principle can be applied for transplant recipients with isolated draining biliary ducts. Percutaneous therapy of isolated draining biliary ducts with absolute alcohol (ie, 100% ethanol) was successfully performed in a 7-year-old patient who had undergone orthotopic liver transplantation. The increasing use of segmental living and split cadaveric liver grafts, with the subsequent increase risk in biliary complications, necessitates more efficient therapy for isolated draining bile ducts. BILIARY complications commonly seen after pediatric liver transplantation include anastomotic leakage, anastomotic stenosis with bile duct dilation, nonanastomotic strictures from hepatic arterial insufficiency, intrahepatic bile duct stones, sludge or debris, and biloma (1Egawa H. Uemoto S. Inomata Y. et al.Biliary complications in pediatric living related liver transplantation.Surgery. 1998; 124: 901-910Abstract Full Text Full Text PDF PubMed Scopus (104) Google Scholar, 2Thuluvath P.J. Pfau P.R. Kimmey M.B. et al.Biliary complications after liver transplantation: the role of endoscopy.Endoscopy. 2005; 37: 857-863Crossref PubMed Scopus (170) Google Scholar, 3Lopez-Santamaria M. Martinez L. Hierro L. et al.Late biliary complications in pediatric liver transplantation.J Pediatr Surg. 1999; 34: 316-320Abstract Full Text PDF PubMed Scopus (45) Google Scholar, 4Lallier M. St-Vil D. Luks F.I. et al.Biliary tract complications in pediatric orthotopic liver transplantation.J Pediatr Surg. 1993; 28: 1102-1105Abstract Full Text PDF PubMed Scopus (31) Google Scholar). This report describes the successful use of alcohol sclerotherapy for occlusion of isolated draining biliary ducts that had been conservatively managed with external biliary drainage for several years. The patient is a 7-year-old boy who underwent orthotopic liver transplantation at the age of 30 months for end-stage liver disease secondary to biliary atresia and a failed Kasai portoenterostomy procedure. Transplantation was performed using an ABO-identical split liver graft (segments II and III) via a piggyback technique in which the recipient’s inferior vena cava was anastomosed to the graft hepatic vein. The hepatic artery was reconstructed with a microsurgical technique. Biliary reconstruction was performed using a left hepaticojejunostomy with a Roux-en-Y limb anastomosis. The course after transplantation was complicated by hepatic artery thrombosis, which was confirmed by Doppler ultrasonography of the liver on postoperative day 25. Contrast-enhanced computed tomography (CT) of the abdomen and pelvis on postoperative day 26 demonstrated dilation of the central right-sided biliary ducts and infarction of liver parenchyma at the most lateral region of the left side of the liver graft (Fig 1). At this time, the liver graft function test results were all increased, with an alanine aminotransferase level of 618 U/L (normal range, 5–50 U/L), total bilirubin level of 2.9 mg/dL (normal range, 0.2–1.5 mg/dL), alkaline phosphatase of 301 U/L (range, 35–110 U/L), bilirubin level of 1.9 mg/dL (normal range, 0.1–0.4 mg/dL), and International Normalized Ratio of 1.3. Urgent repeat transplantation was not indicated, but there was clinical concern that biliary tract obstruction was present. A percutaneous transhepatic cholangiogram was obtained in the interventional radiology unit on postoperative day 29. The initial cholangiogram demonstrated diffusely dilated central biliary ducts with no significant peripheral biliary dilation. Injection of the superior portion of the biliary tree demonstrated a rounded collection of contrast medium lateral to the biliary ducts consistent with a biloma. Although the biloma was not seen on the recent CT study, it may have been masked by the hypoenhancing region of infarcted liver graft. The small bowel was not opacified. The Roux-en-Y left hepaticojejunostomy anastomotic site was tightly stenotic, and attempts at crossing the anastomosis were unsuccessful despite attempted access at two different sites. Two 6-F external biliary drains (UreSil, Skokie, Ill) were placed in a superior and inferior duct, respectively, to allow decompression of the biliary tree. No other strictures were identified. A second cholangiogram obtained 1 week later demonstrated interval decrease in dilation of the biliary ducts with mild irregularity of the ducts. The superior left-sided drain became nonfunctional and was removed. Subsequently, the anastomosis was crossed. Follow-up cholangiograms demonstrated drainage of the left-sided biliary tree into the anastomosis. However, a new fresh-stick cholangiogram demonstrated an isolated collection of upper right-sided biliary ducts that persisted to drain externally without communication with the remainder of the biliary system or the jejunostomy (Fig 2a). These isolated upper right-sided biliary ducts were conservatively managed with external biliary drainage (UreSil) without closure of the isolated bile ducts and with preservation of good graft function. Sclerotherapy was considered earlier but was not performed as a result of parental concerns regarding the novelty of this treatment, the stable clinical state of the patient, and the normal progressive growth of the remainder of the graft. Four years after orthotopic liver transplantation, serial absolute alcohol (ie, 100% ethanol) sclerotherapy was performed for the final therapy of these isolated upper right-sided biliary ducts. Before the procedure, the alanine aminotransferase level was 28 U/L, total bilirubin level was 0.8 mg/dL, and alkaline phosphatase level was 294 U/L. For each session, under general anesthesia, external biliary catheter exchange was performed followed by sclerotherapy through the newly placed catheter. The patient received prophylactic antibiotics (intravenous ceftriaxone and metronidazole) and was monitored for subsequent pain, fever, infection, abscess formation, and worsening liver function. In the first session, sclerotherapy was initiated with 0.6 mL of absolute alcohol injected through the catheter and left in place for 2 minutes. The alcohol was then aspirated and the catheter was flushed with 1 mL of saline solution. There was no immediate cholangiographic improvement (Fig 2a). However, biliary drainage from those ducts decreased from 15 mL/d to 7 mL per 48 hours. The patient tolerated the procedure well; there was no postprocedural fever, and liver function remained essentially unchanged from baseline with an alanine aminotransferase level of 37 U/L, total bilirubin level of 0.8 mg/dL, and alkaline phosphatase level of 287 U/L. The patient was brought back 6 weeks later for a second session of alcohol sclerotherapy. One milliliter of absolute alcohol was injected through the catheter and left in place for 2 minutes. Another 6 weeks later, the patient was brought back for a third session of alcohol sclerotherapy. Four milliliters of absolute alcohol was instilled into the isolated ducts and allowed to remain for 2 minutes. The residual alcohol was drained. At the final session 7 weeks later, 1 mL of absolute alcohol was instilled into the small residual isolated duct (Fig 2b). The external biliary drainage catheter was finally removed 19 weeks after the initial injection (Fig 2c). The patient has not required any further intervention 15 months after alcohol sclerotherapy. Percutaneous alcohol sclerotherapy with ethanol has been described for the treatment of biliary cutaneous fistulas in patients who have undergone hepatic resection (5Matsumoto T. Iwaki K. Hagino Y. et al.Ethanol injection therapy of an isolated bile duct associated with a biliary-cutaneous fistula.J Gastroenterol Hepatol. 2002; 17: 807-810Crossref PubMed Scopus (38) Google Scholar, 6Kyokane T. Nagino M. Sano T. et al.Ethanol ablation for segmental bile duct leakage after hepatobiliary resection.Surgery. 2002; 131: 111-113Abstract Full Text Full Text PDF PubMed Scopus (37) Google Scholar). Ethanol was initially used for the ablation of renal cysts and is commonly used for treatment of cystic and solid hepatic lesions (7Gelczer R.K. Charboneau J.W. Hussain S. et al.Complications of percutaneous ethanol ablation.J Ultrasound Med. 1998; 17: 531-533PubMed Google Scholar). Park et al (8Park J.H. Oh J.H. Yoon Y. et al.Acetic acid sclerotherapy for treatment of a biliary leak from an isolated bile duct after hepatic surgery.J Vasc Interv Radiol. 2005; 16: 885-888Abstract Full Text Full Text PDF PubMed Scopus (25) Google Scholar) described the successful use of acetic acid for treatment of isolated bile ducts after hepatic surgery. Absolute alcohol (ie, 100% ethanol) was the medium we used for the treatment of the isolated draining biliary ducts in our patient. It has the advantage of being widely available, inexpensive, and easy to administer with a low risk of complications (7Gelczer R.K. Charboneau J.W. Hussain S. et al.Complications of percutaneous ethanol ablation.J Ultrasound Med. 1998; 17: 531-533PubMed Google Scholar). The initial volume of alcohol selected was matched to the amount of iodinated contrast medium that opacified the isolated bile ducts. The procedures were arbitrarily spaced 6–8 weeks apart because that has been a duration of time during which we have seen healing and response to other biliary interventions in liver transplant recipients. We chose to be cautious in this child in whom each procedure required hospital admission and general anesthesia. Further experience is needed to determine whether a shorter interval would be sufficient. Alcohol sclerotherapy is potentially advantageous for use in pediatric patients. Chronic external biliary drainage may not be advantageous for pediatric patients as a result of the multiple sessions of exposure to radiation (19 sessions of external biliary catheter exchanges vs four sessions of sclerotherapy in this case) and the resultant decrease in quality of life. The isolated biliary ducts were treated successfully in this case with only four sessions of alcohol sclerotherapy. The treatment obviated additional biliary procedures and the attendant radiation exposure. The number of pediatric liver transplantations has increased as a result of improved surgical technique, greater availability of size-appropriate donor livers, advances in immunosuppression therapy, and advances in organ preservation (9Abt P.L. Rapaport-Kelz R. Desai N.M. et al.Survival among pediatric liver transplant recipients: impact of segmental grafts.Liver Transpl. 2004; 10: 1287-1293Crossref PubMed Scopus (53) Google Scholar, 10Kim J.S. Broering D.C. Tustas R.Y. et al.Split liver transplantation: past, present and future.Pediatr Transplant. 2004; 8: 644-648Crossref PubMed Scopus (36) Google Scholar, 11Goss J.A. Shackleton C.R. Swenson K. et al.Orthotopic liver transplantation for congenital biliary atresia: an 11-year, single-center experience.Ann Surg. 1996; 224: 276-284Crossref PubMed Scopus (71) Google Scholar). Biliary complications comprise as many as 50% of posttransplantation complications (1Egawa H. Uemoto S. Inomata Y. et al.Biliary complications in pediatric living related liver transplantation.Surgery. 1998; 124: 901-910Abstract Full Text Full Text PDF PubMed Scopus (104) Google Scholar, 2Thuluvath P.J. Pfau P.R. Kimmey M.B. et al.Biliary complications after liver transplantation: the role of endoscopy.Endoscopy. 2005; 37: 857-863Crossref PubMed Scopus (170) Google Scholar). With the increased incidence of pediatric liver transplantation and its potential for biliary complications, interventional radiology has an important role in the care of these patients. In this case, persistent draining isolated bile ducts from a nonanastomotic stricture caused by hepatic arterial insufficiency was definitively and successfully treated with only four sessions of sclerotherapy. Our experience suggests that local administration of alcohol sclerotherapy may be a useful technique in the liver transplant recipient for the management of isolated biliary ducts that fail to respond to conservative management. Further experience is necessary to determine the appropriate interval and frequency of sclerotherapy sessions.