Refractory Intrahepatic Bile Leak in a Young Patient After Blunt Abdominal Trauma: Technique of Endoscopic Management Using Cyanoacrylate Glue-Lipiodol Combination

ABSTRACT

Bile leaks are commonly managed endoscopically with either sphincterotomy and/or biliary stenting. Historically, if these procedures failed, surgery was inevitable, carrying high morbidity. Data are sparse on the management of persistent or refractory bile leaks after failed endoscopic retrograde cholangiopancreatography. We report a young patient, following blunt abdominal trauma, managed with percutaneous catheter drains for bilomas. Ongoing bilious output in the perihepatic drain persisted, despite 3 sessions of biliary stenting and subsequent stent upgradations. Definitive management was achieved by endoscopic injection of glue-lipiodol (1:1) at the leak site under real-time fluoroscopic guidance, highlighting a minimally invasive alternative to surgery.

INTRODUCTION

Bile leaks are commonly reported after hepatobiliary surgeries (3.6%–12%) and abdominal trauma (0.5%–21%).¹ Most leaks resolve spontaneously within 3 weeks if adequate biliary drainage is achieved.¹ High-grade leaks usually present with biliary peritonitis or biloma, abdominal pain, and fever.^2,3 Endoscopic retrograde cholangiopancreatography (ERCP) using endoscopic sphincterotomy (EST), biliary stenting, or nasobiliary drain placement is usually the preferred treatment modality. It decreases the transpapillary gradient and diverts bile into the duodenum.^4–6 Clinical success is reported to be around 87%–100%.^3,7–10 Nevertheless, refractory bile leaks are challenging to treat, and evidence regarding optimal rescue therapy is limited. Few approaches such as fully covered self-expanding metal stents (FC-SEMS) or use of coil with/without n-butyl-2-cyanoacrylate (NBCA) glue combination have been reported in isolated case reports.^7,8,11–13 In this study, we describe a case of a refractory high-grade peripheral leak following blunt abdominal trauma managed successfully using NBCA glue-lipiodol combination.

CASE REPORT

A 14-year-old adolescent boy presented with abdominal pain and vomiting following blunt abdominal trauma in a road-side accident. On examination, he had tachycardia (130/minute) but normotensive. Blood investigations revealed severe anemia (hemoglobin 5.6 g/dL; Table 1). After resuscitation with blood transfusions, intravenous fluids, and antibiotics, contrast-enhanced computed tomography of the abdomen with angiography was performed, which revealed grade IV liver injury, right hepatic artery pseudoaneurysm, grade II renal injury, and moderate hemoperitoneum. Interventional radiology-guided coil embolization of the right hepatic artery pseudoaneurysm was successfully performed (Figure 1).

Table 1.

Laboratory investigations of the patient at the time of presentation to the emergency

Parameters	Results
Hemoglobin	5.6 g/dL
TLC	8,800/mm3
Platelet	2,40,000/mm3
Urea	18 mg/dL
Creatinine	0.6 mg/dL
Total bilirubin	1.2 mg/dL
Direct bilirubin	0.4 mg/dL
SGOT	80 IU/L
SGPT	102 IU/L
ALP	114 IU/L
Total protein	6.6 g/dL
Albumin	3.2 g/dL
PT	11
APTT	31
INR	1.1
HIV	Not reactive
HBsAg	Not reactive
Anti-HCV	Not reactive
Fasting blood sugar	82 mg/dL
C-reactive protein	67 mg/dL

ALP, alkaline phosphatase; APTT, activated partial thromboplastin time; HBsAg, hepatitis B surface antigen; HCV, hepatitis C virus; INR international normalised ratio; PT, prothrombin time; SGOT, serum glutamic oxaloacetic transaminase; SGPT, serum glutamic oxaloacetic transaminase; TLC, total leucocyte count.

Figure 1.

(A) Contrast-enhanced computed tomography abdomen revealed grade IV liver injury (multiple linear confluent nonenhancing lacerations are seen in segment V–VIII of the right lobe), traversing through the liver capsule (yellow solid arrow), along with hemoperitoneum. (B) CT angiography revealed an eccentric saccular outpouching (6.2 × 3.5 mm) from a branch of RHA pseudoaneurysm (yellow solid arrow). (C) Coil-artifacts (yellow solid arrow) noted in RHA after digital subtraction angiography coil embolization. (D) Percutaneous catheter drain (10Fr; yellow solid arrow) noted in pelvic collection (biloma; orange asterisk). RHA, right hepatic artery.

Subsequently, percutaneous catheter drainage (PCD; 10 Fr) was placed in the pelvic hypodense collection draining bilious output (aspirate bilirubin 30 mg/dL; Figure 1). As a result, ERCP was performed, which revealed a high-grade leak from the right hepatic ductal system, and 7Fr × 10 cm double pigtail (DPT) stent was placed (Figure 2). Because of persistent fever and abdominal pain, 2 additional PCDs (10Fr) were placed in peri-hepatic and pelvic collections and antibiotics were upgraded. The pelvic collections subsequently resolved, and PCDs were removed. However, persistent bilious drainage from the perihepatic PCD prompted a second ERCP, and the stent was upgraded to 10Fr × 10 cm DPT (Figure 2). Despite this, the patient continued to have intermittent fever and purulent bilious output (160–200 mL/d). A repeat contrast-enhanced computed tomography of the abdomen revealed a residual perihepatic collection (5 × 2 × 7.4 cm), and PCD was upgraded to 14 Fr. A third ERCP was performed because of ongoing bilious output. Selective cannulation of the right-anterior (hepatic) peripheral duct (RAPD) revealed a high-grade leak from one of the peripheral branches. A 7Fr × 15 cm DPT was placed till the mouth of the visualized leak (Figure 3). However, 2 weeks later, bilious output still persisted (150 mL/d).

Figure 2.

(A) Cholangiogram showed a high-grade leak from the right hepatic ductal system (yellow solid arrow). (B) A 7Fr × 10 cm DPT stent was placed (yellow solid arrow) (Note: In [A] and [B], previously placed coils in RHA pseudoaneurysm noted in fluoroscopic image [orange dotted arrow]). (C) Cholangiogram showed a high-grade leak from the right hepatic ductal system (yellow solid arrow). (D) A 10Fr × 10 cm DPT stent was placed (yellow solid arrow) (Note: In [C] and [D], previously placed coils in RHA pseudoaneurysm [orange dotted arrow] and right perihepatic percutaneous catheter drain noted in fluoroscopic image [white dotted arrow]). DPT, double pigtail; RHA, right hepatic artery.

Figure 3.

(A) Cholangiogram revealed extravasation of contrast from the right ductal system (yellow solid arrow). (B) Selective cannulation of the right-anterior (hepatic) peripheral duct revealed a high-grade leak from one of the peripheral branches. (C) A 7Fr × 15 cm double pigtail stent was placed till the mouth of the visualized leak (yellow solid arrow) (Note: In [A–C], previously placed coils in right hepatic artery pseudoaneurysm [orange dotted arrow] and right perihepatic percutaneous catheter drain [white dotted arrow] noted in fluoroscopic image).

After failure of 3 sequential ERCP-guided stenting sessions, a multidisciplinary team decided on using a combination of NBCA glue with lipiodol to seal the peripheral leak. Under real-time fluoroscopic guidance, an ERCP cannula (Tandem XL Triple-Lumen, Boston Scientific) was used to cannulate the selective branch of the RAPD and contrast was injected, which demonstrated the extravasation of the contrast into a small cavity and subsequent free leak into the subhepatic space. A combination of 0.5 mL NBCA glue with equal amounts of lipiodol (0.5 mL) in a 1:1 ratio was used and injected at the site of the disruption using the cannula. Repeat cholangiogram confirmed obliteration of the leak (Figure 4; Video 1). Within 48 hours, drain output ceased completely, perihepatic PCD was removed, and the patient was discharged. Repeat computed tomography after 1 month revealed complete resolution of the collection and glue-lipiodol cast at the treatment site (Figure 5). The patient followed up in the outpatient (OPD) services of the surgery and gastroenterology department for a year, with no fresh complaints. Figure 6 details the whole timeline of the index case.

Figure 4.

(A) Cholangiogram shows an ERCP cannula which was used to cannulate the selective branch of the RAPD (yellow solid arrow) and contrast was injected which demonstrated the extravasation of the contrast into a small cavity (orange dotted arrow) and subsequent free leak into the subhepatic space (white dotted arrow). (B) Zoom image (×10 times) shows the contrast extravasation into the cavity (orange dotted arrow) [Note: Yellow solid arrow refers to the radio-opaque tip of the ERCP cannula in the RAPD]. (C) A combination of 0.5 mL n-butyl-2-cyanoacrylate glue with equal amounts of lipiodol was used and injected at the site of the disruption using the cannula; bubbles (orange dotted arrow) of the combination were noted inside the cavity. (D) Repeat cholangiogram taken revealed complete obliteration of the leak (no passage of contrast into the cavity and subhepatic space) (Note: Previously placed coils in right hepatic artery pseudoaneurysm and right perihepatic percutaneous catheter drain noted in fluoroscopic image). ERCP, endoscopic retrograde cholangiopancreatography; RAPD, right-anterior (hepatic) peripheral duct.

Figure 5.

(A) X-ray abdomen performed after intervention showed a radio-opaque glue-lipiodol complex (yellow solid arrow) at the site of injection. Also, the right peri-hepatic percutaneous catheter drain (white dotted arrow) and previously placed coils in the right hepatic artery (orange dotted arrow) were visualized. (B) Contrast-enhanced computed tomography of the abdomen (1-month follow-up) revealed no collection and the presence of lipiodol-glue complex (yellow solid arrow) at the site of injection.

Figure 6.

Timeline of the index case detailing the initial trauma, percutaneous catheter drainage placements, various endoscopic retrograde cholangiopancreatography sessions, and final n-butyl-2-cyanoacrylate glue-lipiodol session. DPT, double pigtail; ERCP, endoscopic retrograde cholangiopancreatography; NBCA, n-butyl-2-cyanoacrylate; PCD, percutaneous catheter drain; RAPD, right-anterior (hepatic) peripheral duct.

DISCUSSION

Endoscopic management using ERCP is the standard first-line treatment for the management of bile leaks. It decreases the transpapillary pressure gradient and promotes preferential bile flow in the duodenum, rather than extravasation at the leak site. While most leaks are managed using EST and/or stenting, refractory leaks remain a therapeutic challenge. They have been defined as a leak that fails to close after 2 weeks of endoscopic intervention with a combination of EST and a 10Fr transpapillary biliary stent, regardless of the biliary leak location.¹¹ High-grade leaks from peripheral duct are often difficult to tackle and may sometimes warrant surgical intervention.^1,9 For management of refractory or complex peripheral bile leaks, various novel, innovative endoscopic modalities have been proposed, which have been described in isolated case reports or case series, such as coil, NBCA glue, fibrin glue, FC-SEMS, or a combination of the same. Seewald et al¹² reported the first use of NBCA glue in 9 cases of bile leak, in which the initial endoscopic treatment was unsuccessful, and clinical success was achieved in 7 cases. Kahaleh et al⁷ described the use of FC-SEMS in 16 cases of bile leak. In our index case, the key challenge was to locate the selective peripheral anterior branch leak, cannulate it, and use minimal glue (0.5 mL) to effectively seal the leak. Our follow-up imaging clearly shows the final location of the glue cast just beneath the liver capsule (Figure 5).

Our case is similar to few cases published in the literature regarding the use of cyanoacrylate glue for sealing the bile leak. The case by Pulliam et al¹⁴ used specialized microcatheter and microwires to reach the selective leaking duct for the glue injection, which could be a more expensive strategy; we were able to achieve the same using our regular day cannula (as noted in the image; Figure 4). The 2 cases described by Patel et al¹⁵ again had used a costlier choledochoscope to guide the glue injection. Thus, our index case, while presenting as a therapeutic challenge, was unique in its approach that it could be managed effectively with precision targeting using a simple regular use cannula without the need for microcatheters or choledochoscope.

Our case adds to the limited data on the effective use of such agents for endoscopic management of refractory bile leaks. NBCA glue polymerizes on contact with biological tissue, arresting the leak by plugging fistulous tracts arising from the bile duct lumen. Another “pro” is its immediate effect, which precludes the need for successive procedures and prolonged hospitalization. On the other hand, lipiodol serves 2 important advantages: (i) delays glue polymerization making it easier to inject and preventing it from solidifying inside the catheter and (ii) being radiopaque, it ensures optimum delivery of the glue to the desired site under fluoroscopic guidance.

Hence, key points to prevent nontarget embolization and other adverse events in the procedure were as follows: (i) locate and cannulate the selective peripheral anterior branch leak and use minimal glue (0.5 mL) to effectively seal the leak. This ensured nontarget embolization. We specifically aimed at achieving this, to avoid obliterating any major biliary ductal branch in a such a young patient; (ii) the choice of dilution has been selectively chosen to optimize the polymerization; (iii) minimal glue (0.5 mL) was used (much lower than previously described—1–1.5 mL), as it was perceived that the leak site was small and from a peripheral duct. This avoided inadvertent spillage of glue beyond the target; (iv) the whole procedure of NBCA: Lipiodol injection was performed under constant fluoroscopic guidance to ensure precise and optimum delivery of the mixture to the leak site and subsequent contrast injection was used to document real time “sealing” of the leak; and (v) peri-procedure antibiotics were administered and the PCD output was monitored for the next 48 hours for drainage of residual fluid.

Hence, the use of NBCA glue-lipiodol combination through ERCP for sealing off refractory bile leak highlights that this occlusion is a safe, effective, and minimally invasive alternative for control of refractory bile leaks precluding surgical intervention. Considering its positive outcomes, larger, prospective data are needed to validate its safety and efficacy on a broader scale and further support its application in our endoscopy practice.

DISCLOSURES

Author contributions: J. Dhar: patient management, first draft of the article, acquisition, analysis and interpretation of data, final approval of the manuscript; CL Birda, A. Kumar, PB Neelam, U. Gorsi, C. Tandup, R. Kochhar: patient management, acquisition of data, analysis and interpretation of data, critical revision of the manuscript, final approval of the manuscript; J. Samanta: conception and design, patient management, performed the procedure, acquisition of data, analysis and interpretation of data, critical revision of the manuscript, final approval of the manuscript. All authors agree and approve the final version of the manuscript. J. Samanta is the article guarantor.

Financial disclosure: None to report.

Informed consent was obtained for this case report.

ABBREVIATIONS:

ALP

alkaline phosphatase

APTT

activated partial thromboplastin time

DPT

double pigtail

ERCP

endoscopic retrograde cholangiopancreatography

EST

endoscopic sphincterotomy

FC-SEMS

fully covered self-expanding metal stents

HBsAg

hepatitis B surface antigen

HCV

hepatitis C virus

INR

international normalised ratio

NBCA

n-butyl-2-cyanoacrylate

PCD

percutaneous catheter drain

PT

prothrombin time

RAPD

right-anterior (hepatic) peripheral duct

RHA

right hepatic artery

SGOT

serum glutamic oxaloacetic transaminase

TLC

total leucocyte count