- Email: [email protected]
Role of ERCP in the management of non-iatrogenic traumatic bile duct injuries in the pediatric population
CASE STUDIES
Role of ERCP in the management of non-iatrogenic traumatic bile duct injuries in the pediatric population Alex Ulitsky, MD, Steven Werlin, MD, Kulwinder S. Dua, MD Milwaukee, Wisconsin, USA
Iatrogenic biliary injuries are recognized postoperative complications of cholecystectomy and other hepatobiliary surgeries. Management of these injuries often involves surgical, radiologic, and endoscopic interventions. Endotherapy in the form of biliary sphincterotomy, transpapillary stenting, or both are accepted interventions for these patients.1,2 Non-iatrogenic injuries to the bile ducts from abdominal trauma can be a source of significant short-term and long-term morbidity. Many of these patients undergo surgery for multiple-organ injuries, and ongoing bile leaks often lead to surgical reintervention. Therapy for biliary tract injuries in these patients can be challenging and complicated because of accompanying multiple-organ trauma and infection. There is no consensus on the treatment of noniatrogenic traumatic bile leaks, and decisions are often based on the extent and mechanism of injury, associated organ injuries, and local expertise. We and others have previously reported on the successful management of traumatic bile duct leaks in adults by ERCP with biliary sphincterotomy and stent placement.3,4 However, there is limited published information on the role of ERCP in treating traumatic biliary injuries in children. The only case series includes 5 children,5 whereas others have published case reports.6-9 We, therefore, reviewed our experience with endoscopic management of biliary tract injuries resulting from abdominal trauma in children.
Abbreviations: ES, endoscopic sphincterotomy.
PATIENTS AND METHODS At a single, tertiary-care medical center, children with complex bile duct injuries were referred for ERCP by the surgical trauma service for the management of bile leakage after blunt or sharp abdominal trauma. The data were prospectively collected and included demographics, nature of trauma, type and location of bile duct injury, details of therapeutic intervention performed at ERCP, and final outcome, including timing of leak resolution and any complications. After initial surgical or radiologic interventions, patients with continuing bile leakage, defined as bilious output from percutaneous drains or radiologic evidence of biloma, were referred for ERCP, to diagnose the site of bile leakage. Therapeutic interventions (endoscopic sphincterotomy [ES] with or without transpapillary stent placement) were performed during ERCP. Biliary stenting was performed with 5F or 10F plastic biliary stents. Bile leak resolution was defined as stoppage of bilious output from abdominal or chest drains, with normalization of liver function test results. Resolution of leakage was further confirmed by a repeat ERCP 6 to 8 weeks later, during which the previously placed transpapillary stents were removed. All ERCPs and therapeutic interventions were performed by one senior faculty member (K.D.). The Human Research Review Committee of the Medical College of Wisconsin and the Human Rights Review Board of the Children’s Hospital of Wisconsin approved this study.
DISCLOSURE: All authors disclosed no financial relationships relevant to this publication.
RESULTS
Copyright © 2011 by the American Society for Gastrointestinal Endoscopy 0016-5107/$36.00 doi:10.1016/j.gie.2010.11.054
Eight consecutive children with traumatic bile duct injuries and bile leakage were reviewed in this case series (Table 1). The patients were 5 boys and 3 girls, with a mean age of 12.5 years (range 3-17 years). Seven of the children sustained biliary injury from blunt abdominal trauma: motor vehicle accident (4), fall from a height (2), and injury by a school bus (1). One sustained a penetrating bile duct injury from a gunshot. Five (63%) underwent surgical intervention before ERCP for multiple-organ injury, during which primary repair of the bile ducts was not performed. All the patients had abdominal drains placed
Division of Gastroenterology and Hepatology (A.U., K.S.D.), Froedtert Memorial Hospital, Medical College of Wisconsin, Division of Gastroenterology and Hepatology (S.W.), Children’s Hospital of Wisconsin, Medical College of Wisconsin, Milwaukee, Wisconsin. Presented at Digestive Disease Week, May 1-6, 2010, New Orleans, Louisiana (Gastrointest Endosc 2010;71:AB109). Reprint requests: Kulwinder S. Dua, MD, Division of Gastroenterology and Hepatology, Medical College of Wisconsin, 9200 West Wisconsin Avenue, Milwaukee, WI 53226.
www.giejournal.org
Volume 73, No. 4 : 2011 GASTROINTESTINAL ENDOSCOPY 823
ERCP in pediatric bile duct injuries
Ulitsky et al
TABLE 1. Patient characteristics and interventions performed to treat biliary leaks
Patient no.
Age, y
Sex
Injury
Intervention before ERCP
Location of bile leak
Intervention during ERCP
Days to leak resolution
1
14
M
Fall from height
Drain
Right intrahepatic
1: ES 2: ES and stent 3: Stent removal
20*
2
3
M
Hit by car
Ex-lap; drain
Right intrahepatic
1: ES and stent 2: Stent removal
10
3
17
F
MVC
Ex-lap; drain
Left intrahepatic
1: ES and stent 2: Stent removal
5
4
16
F
MVC
Drain
Hilar and left intrahepatic
1: ES and stent 2: Stent removal
3
5
10
M
Hit by car
Ex-lap; drain
Hilar
1: ES and stent 2: Stent removal
10
6
15
F
Fall from horse
Ex-lap; drain
Right intrahepatic
1: ES and stent 2: Stent removal
15
7
6
M
Run over by bus
Ex-lap; drain
Left intrahepatic
1: ES and stent 2: Stent exchange 3: Stent removal
10*
8
16
M
GSW
Drain
Right intrahepatic
1: ES and stent 2: Stent removal
20
M, Male; ES, endoscopic sphincterotomy; Ex-lap, exploratory laparotomy; F, female; MVC, motor vehicle crash; GSW, gunshot wound. *Days after 2nd ERCP.
Figure 1. Representative patient 2 (age 3 years) who was hit by a car and sustained multiple-organ trauma. A, Right intrahepatic bile leak (arrow) managed by ERCP sphincterotomy and biliary stent placement. The leak resolved in 10 days, as determined by absent output from the percutaneous drain and imaging studies showing no biloma. B, Follow-up ERCP to remove the stent confirmed resolution of the leak.
824 GASTROINTESTINAL ENDOSCOPY Volume 73, No. 4 : 2011
www.giejournal.org
Ulitsky et al
either during the exploratory laparotomy or as a separate procedure by interventional radiology. ERCP was successful for all of the patients (representative patient 2, Fig. 1). The endoscopes used were Olympus (Olympus America, Center Valley, Penn) PJF160 (diameter 7.5 mm; patients 2 and 7), JF140 (diameter 11.0 mm; patient 5), and TJF160VF (diameter 11.3 mm; patients 1, 3, 4, 6, and 8). No technical difficulties were encountered in any patient because of altered anatomy, fresh surgical wounds, or other coexisting conditions. All patients were given general anesthesia by the pediatric anesthesiologist and were intubated. The median time for ERCP was 42 minutes (range 28-74 minutes). Seven patients had injury to the intrahepatic bile ducts, 3 in the left hepatic lobe and 4 in the right lobe. One patient had an isolated hilar (extrahepatic) leak, and 1 patient had both a hilar and an intrahepatic injury. The first patient in the series had ES alone performed, which was followed by brisk drainage of contrast material and bile. However, after the procedure, the percutaneous biliary output increased, probably secondary to papillary edema after sphincterotomy. A repeat ERCP was performed 6 days later, and a biliary stent was placed, which led to the resolution of the leak. All subsequent patients were treated with ES followed by stent placement. Thus, all 8 patients underwent biliary stenting. Bile leaks resolved in all 8 patients. The mean time to resolution was 12 days (range 3-20 days). Patient 7 developed severe GI bleeding and elevation in liver function test results 8 days after the initial ERCP. EGD findings were consistent with hemobilia, leading to clotting of the biliary stent and continued bile leakage. The sphincterotomy site was not bleeding. Hence, an angiography was performed that identified the leak from a small intrahepatic branch of the hepatic artery. After successful hepatic artery branch embolization, the patient had a repeat ERCP (Fig. 2). On ERCP, the previously placed 5F plastic biliary stent was found to be occluded with blood clots and was replaced with two 5F plastic stents. Two 5F stents were used instead of a 10F stent because a pediatric duodenoscope (Olympus) that allows only 5F accessories was used for the procedure. The leak resolved 10 days after the second ERCP. The cholangiogram performed during stent removal 54 days after the second ERCP showed a cut-off of the left intrahepatic duct without further leakage, and the stents were removed. Imaging studies showed an atrophic left lobe, and because the liver blood test result remained normal, no further surgical intervention was considered. One patient (patient 8) developed a biliary stricture at the site of the leak, which was identified 5 months after the injury when the patient presented with abdominal discomfort, abnormal liver test results, and dilated right intrahepatic ducts on imaging studies. Although endoscopic management may have been suitable for www.giejournal.org
ERCP in pediatric bile duct injuries
this stricture, the family of the patient and the referring surgeon opted for a surgical approach as against repeated endoscopic interventions. No immediate or late complications of ERCP were observed in any of the patients.
DISCUSSION In this review, we report the results for 8 children treated endoscopically for abdominal trauma–related biliary injuries. Most of the patients initially had undergone surgery for multiple intra-abdominal organ injuries and continued to have bile leaks postoperatively. Endoscopic management of iatrogenic bile-duct injuries associated with hepatobiliary surgeries (eg, postcholecystectomy leaks and strictures) is well-established in the adult literature.1,2 The procedures include ES and/or stenting for biliary leaks as well as dilation and stenting for strictures. Non-iatrogenic injuries in the form of blunt and sharp trauma to the abdomen also can result in bile duct injuries because the liver is the most common organ affected with abdominal trauma. These injuries can be a significant source of morbidity because they are frequently associated with other organ trauma and infection. Surgical treatment, especially reoperation to manage biliary injuries, is difficult and can be associated with high morbidity including complications such as stricture formation, further leaks, and cholangitis. Endoscopic treatment of non-iatrogenic, traumatic bile duct injuries, however, may be as effective as treatment of iatrogenic injuries. Similar to iatrogenic postoperative bile leaks, case series on adults have also shown that ERCP with biliary stenting and/or ES could help in resolving bile leaks secondary to non-iatrogenic trauma.3,4 The pediatric literature on this topic is limited, with only a few case reports and one small case series.5-9 In their series of 5 patients, Castignetti et al5 demonstrated the safety and effectiveness of ERCP with biliary stenting and sphincterotomy in the noninvasive management of traumatic bile duct injuries. Their series included 5 children aged 10 years or over. Hence, the only type of endoscope used in their series was the Olympus therapeutic TJF 240 endoscope (11-mm diameter). In the current series, there were 2 cases, involving children aged 3 and 6 years, respectively, in which the PJF pediatric endoscope (7.5-mm diameter), which allows 5F accessories and stents, was used. All children in the series by Castignetti et al5 had blunt abdominal trauma with only intrahepatic bile leaks, and 60% did not require surgical intervention for multiple-organ trauma. In the present series, there was a child with a penetrating gunshot injury and another child who was run over by a school bus. The majority of the children in the current series required pre-ERCP surgical interventions for multiple-organ injuries. Despite these differences, we have shown that, irrespective of the child’s age or the nature and severity of the Volume 73, No. 4 : 2011 GASTROINTESTINAL ENDOSCOPY 825
ERCP in pediatric bile duct injuries
Ulitsky et al
Figure 2. A, Left hepatic bile leak (arrow) in patient 7 treated with sphincterotomy and placement of a 5F plastic stent. B, Hemobilia from a bleeding left hepatic artery branch was embolized 8 days later (arrowhead), and a previously placed 5F plastic biliary stent was replaced with two 5F plastic stents (block arrow). C, Follow-up ERCP showed no leakage, but there was stricturing with cut-off of the left hepatic duct (arrow).
injury, endoscopic biliary intervention is feasible, safe, and effective in closing bile leaks secondary to trauma. This study did not address the issue of whether sphincterotomy alone or in combination with stenting is required to achieve closure of intrahepatic leaks. With 8 children, the study would not have been powered enough to make this determination if the patients were randomized. In the literature on the management of iatrogenic bile leaks, the results of sphincterotomy alone, biliary stenting alone, or the combination of the two remains controversial. In our previously published series on adult patients with traumatic bile duct injuries, 7 of 8 patients underwent sphincterotomy with stenting; however, 1 patient did well with sphincterotomy alone.3 In a study of 207 adults with postcholecystectomy bile leaks, Sandha et al2 classified bile leaks as low grade (leak identified only after opacification 826 GASTROINTESTINAL ENDOSCOPY Volume 73, No. 4 : 2011
of intrahepatic ducts) or high grade (leak identified before opacification of intrahepatic ducts). Seventy-five patients with low-grade bile leaks underwent sphincterotomy alone, with successful closure of leaks in 91% of cases. The high-grade leak group had stent placement, with a 100% success rate in sealing the bile leak. In this study, there were 2 children with high-grade leaks (patients 4 and 5) and 6 with low-grade leaks. Because our first patient had a low-grade intrahepatic bile leak, we performed sphincterotomy alone in that case. Although leaks may take several days to close, in this patient the percutaneous biliary output increased after the sphincterotomy, probably secondary to postprocedure papillary edema. Hence, we brought the patient back and placed a transpapillary stent. Although no firm conclusions can be made for this patient, we elected to perform sphincterotomy with stent www.giejournal.org
Ulitsky et al
ERCP in pediatric bile duct injuries
multiple-organ trauma, as with the management of iatrogenic biliary injury, continuing bile leakage in these patients can be managed effectively and safely by using therapeutic ERCP.
placement in all subsequent patients. However, because long-term consequences of sphincterotomy in children are not known, transpapillary stenting alone may have been an attractive alternative, and this could be a subject of investigation in a prospective, multicenter, randomized study. The majority of our patients had undergone extensive surgical exploration and therapy for treatment of multiple non-biliary organ injuries before referral for ERCP. In these patients, biliary tree injuries were recognized by the bilious fluid in abdominal or chest drains or by identification of a biloma. Patients were referred for ERCP after biliary leakage continued despite the initial surgical approach. As a result of the therapeutic ERCP intervention, none of the patients required surgical repair of bile ducts to resolve bile leaks. One patient ultimately underwent surgical repair of a biliary stricture. This patient’s family preferred surgery over endoscopic therapy that may have required several procedures. The mean time for leak resolution, as defined by cessation of bilious output from abdominal or chest drains, was 12 days (range 3-20 days) after the ERCP intervention. These outcomes are similar to outcomes achieved for iatrogenic bile leaks by using ERCP interventions.3,4 In summary, as demonstrated in adults, ERCP is a safe and efficacious technique for managing biliary injuries with leaks resulting from blunt or sharp abdominal trauma in children. The use of ERCP was not associated with a higher risk of complications in children with such injuries. Although the majority of patients will undergo surgery for
REFERENCES 1. Bhattacharjya S, Puleston J, Davidson BR, et al. Outcome of early endoscopic biliary drainage in the management of bile leaks after hepatic resection. Gastrointest Endosc 2003;57:526-30. 2. Sandha GS, Bourke MJ, Haber GB, et al. Endoscopic therapy for bile leak based on a new classification: results in 207 patients. Gastrointest Endosc 2004;60:567-74. 3. Bajaj JS, Spinelli KS, Dua KS. Postoperative management of noniatrogenic traumatic bile duct injuries: role of endoscopic retrograde cholangiopancreatography. Surg Endosc 2006;20:974-7. 4. Sharma BC, Mishra SR, Kumar R, et al. Endoscopic management of bile leaks after blunt abdominal trauma. J Gastroenterol Hepatol 2009:24; 757-61. 5. Castignetti M, Houbena C, Patel S, et al. Minimally invasive management of bile leaks after blunt liver trauma in children. J Pediatr Surg 2006;41: 1539-44. 6. Keil R, Snajdauf J, Stuj J, et al. Nonoperative therapy of the posttraumatic biliary fistula in adolescents. Eur J Pediatr Surg 2001;11:274-6. 7. Sharpe RS, Nance ML, Stafford PW. Nonoperative management of blunt extrahepatic biliary duct transection in the pediatric patient: case report and review of the literature. J Pediatr Surg 2002;37:1612-6. 8. Church NG, May G, Sigalet DL. A minimally invasive approach to bile duct injury after blunt liver trauma in pediatric patients. J Pediatr Surg 2002; 37:773-5. 9. Roche B, Mentha G, Bugmann P, et al. Intrahepatic biliary lesions following blunt liver trauma in children: is nonoperative management or conservative operative treatment always safe? Eur J Pediatr Surg 1993;3: 209-12.
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www.giejournal.org
Volume 73, No. 4 : 2011 GASTROINTESTINAL ENDOSCOPY 827
Role of ERCP in the management of non-iatrogenic traumatic bile duct injuries in the pediatric population Alex Ulitsky, MD, Steven Werlin, MD, Kulwinder S. Dua, MD Milwaukee, Wisconsin, USA
Iatrogenic biliary injuries are recognized postoperative complications of cholecystectomy and other hepatobiliary surgeries. Management of these injuries often involves surgical, radiologic, and endoscopic interventions. Endotherapy in the form of biliary sphincterotomy, transpapillary stenting, or both are accepted interventions for these patients.1,2 Non-iatrogenic injuries to the bile ducts from abdominal trauma can be a source of significant short-term and long-term morbidity. Many of these patients undergo surgery for multiple-organ injuries, and ongoing bile leaks often lead to surgical reintervention. Therapy for biliary tract injuries in these patients can be challenging and complicated because of accompanying multiple-organ trauma and infection. There is no consensus on the treatment of noniatrogenic traumatic bile leaks, and decisions are often based on the extent and mechanism of injury, associated organ injuries, and local expertise. We and others have previously reported on the successful management of traumatic bile duct leaks in adults by ERCP with biliary sphincterotomy and stent placement.3,4 However, there is limited published information on the role of ERCP in treating traumatic biliary injuries in children. The only case series includes 5 children,5 whereas others have published case reports.6-9 We, therefore, reviewed our experience with endoscopic management of biliary tract injuries resulting from abdominal trauma in children.
Abbreviations: ES, endoscopic sphincterotomy.
PATIENTS AND METHODS At a single, tertiary-care medical center, children with complex bile duct injuries were referred for ERCP by the surgical trauma service for the management of bile leakage after blunt or sharp abdominal trauma. The data were prospectively collected and included demographics, nature of trauma, type and location of bile duct injury, details of therapeutic intervention performed at ERCP, and final outcome, including timing of leak resolution and any complications. After initial surgical or radiologic interventions, patients with continuing bile leakage, defined as bilious output from percutaneous drains or radiologic evidence of biloma, were referred for ERCP, to diagnose the site of bile leakage. Therapeutic interventions (endoscopic sphincterotomy [ES] with or without transpapillary stent placement) were performed during ERCP. Biliary stenting was performed with 5F or 10F plastic biliary stents. Bile leak resolution was defined as stoppage of bilious output from abdominal or chest drains, with normalization of liver function test results. Resolution of leakage was further confirmed by a repeat ERCP 6 to 8 weeks later, during which the previously placed transpapillary stents were removed. All ERCPs and therapeutic interventions were performed by one senior faculty member (K.D.). The Human Research Review Committee of the Medical College of Wisconsin and the Human Rights Review Board of the Children’s Hospital of Wisconsin approved this study.
DISCLOSURE: All authors disclosed no financial relationships relevant to this publication.
RESULTS
Copyright © 2011 by the American Society for Gastrointestinal Endoscopy 0016-5107/$36.00 doi:10.1016/j.gie.2010.11.054
Eight consecutive children with traumatic bile duct injuries and bile leakage were reviewed in this case series (Table 1). The patients were 5 boys and 3 girls, with a mean age of 12.5 years (range 3-17 years). Seven of the children sustained biliary injury from blunt abdominal trauma: motor vehicle accident (4), fall from a height (2), and injury by a school bus (1). One sustained a penetrating bile duct injury from a gunshot. Five (63%) underwent surgical intervention before ERCP for multiple-organ injury, during which primary repair of the bile ducts was not performed. All the patients had abdominal drains placed
Division of Gastroenterology and Hepatology (A.U., K.S.D.), Froedtert Memorial Hospital, Medical College of Wisconsin, Division of Gastroenterology and Hepatology (S.W.), Children’s Hospital of Wisconsin, Medical College of Wisconsin, Milwaukee, Wisconsin. Presented at Digestive Disease Week, May 1-6, 2010, New Orleans, Louisiana (Gastrointest Endosc 2010;71:AB109). Reprint requests: Kulwinder S. Dua, MD, Division of Gastroenterology and Hepatology, Medical College of Wisconsin, 9200 West Wisconsin Avenue, Milwaukee, WI 53226.
www.giejournal.org
Volume 73, No. 4 : 2011 GASTROINTESTINAL ENDOSCOPY 823
ERCP in pediatric bile duct injuries
Ulitsky et al
TABLE 1. Patient characteristics and interventions performed to treat biliary leaks
Patient no.
Age, y
Sex
Injury
Intervention before ERCP
Location of bile leak
Intervention during ERCP
Days to leak resolution
1
14
M
Fall from height
Drain
Right intrahepatic
1: ES 2: ES and stent 3: Stent removal
20*
2
3
M
Hit by car
Ex-lap; drain
Right intrahepatic
1: ES and stent 2: Stent removal
10
3
17
F
MVC
Ex-lap; drain
Left intrahepatic
1: ES and stent 2: Stent removal
5
4
16
F
MVC
Drain
Hilar and left intrahepatic
1: ES and stent 2: Stent removal
3
5
10
M
Hit by car
Ex-lap; drain
Hilar
1: ES and stent 2: Stent removal
10
6
15
F
Fall from horse
Ex-lap; drain
Right intrahepatic
1: ES and stent 2: Stent removal
15
7
6
M
Run over by bus
Ex-lap; drain
Left intrahepatic
1: ES and stent 2: Stent exchange 3: Stent removal
10*
8
16
M
GSW
Drain
Right intrahepatic
1: ES and stent 2: Stent removal
20
M, Male; ES, endoscopic sphincterotomy; Ex-lap, exploratory laparotomy; F, female; MVC, motor vehicle crash; GSW, gunshot wound. *Days after 2nd ERCP.
Figure 1. Representative patient 2 (age 3 years) who was hit by a car and sustained multiple-organ trauma. A, Right intrahepatic bile leak (arrow) managed by ERCP sphincterotomy and biliary stent placement. The leak resolved in 10 days, as determined by absent output from the percutaneous drain and imaging studies showing no biloma. B, Follow-up ERCP to remove the stent confirmed resolution of the leak.
824 GASTROINTESTINAL ENDOSCOPY Volume 73, No. 4 : 2011
www.giejournal.org
Ulitsky et al
either during the exploratory laparotomy or as a separate procedure by interventional radiology. ERCP was successful for all of the patients (representative patient 2, Fig. 1). The endoscopes used were Olympus (Olympus America, Center Valley, Penn) PJF160 (diameter 7.5 mm; patients 2 and 7), JF140 (diameter 11.0 mm; patient 5), and TJF160VF (diameter 11.3 mm; patients 1, 3, 4, 6, and 8). No technical difficulties were encountered in any patient because of altered anatomy, fresh surgical wounds, or other coexisting conditions. All patients were given general anesthesia by the pediatric anesthesiologist and were intubated. The median time for ERCP was 42 minutes (range 28-74 minutes). Seven patients had injury to the intrahepatic bile ducts, 3 in the left hepatic lobe and 4 in the right lobe. One patient had an isolated hilar (extrahepatic) leak, and 1 patient had both a hilar and an intrahepatic injury. The first patient in the series had ES alone performed, which was followed by brisk drainage of contrast material and bile. However, after the procedure, the percutaneous biliary output increased, probably secondary to papillary edema after sphincterotomy. A repeat ERCP was performed 6 days later, and a biliary stent was placed, which led to the resolution of the leak. All subsequent patients were treated with ES followed by stent placement. Thus, all 8 patients underwent biliary stenting. Bile leaks resolved in all 8 patients. The mean time to resolution was 12 days (range 3-20 days). Patient 7 developed severe GI bleeding and elevation in liver function test results 8 days after the initial ERCP. EGD findings were consistent with hemobilia, leading to clotting of the biliary stent and continued bile leakage. The sphincterotomy site was not bleeding. Hence, an angiography was performed that identified the leak from a small intrahepatic branch of the hepatic artery. After successful hepatic artery branch embolization, the patient had a repeat ERCP (Fig. 2). On ERCP, the previously placed 5F plastic biliary stent was found to be occluded with blood clots and was replaced with two 5F plastic stents. Two 5F stents were used instead of a 10F stent because a pediatric duodenoscope (Olympus) that allows only 5F accessories was used for the procedure. The leak resolved 10 days after the second ERCP. The cholangiogram performed during stent removal 54 days after the second ERCP showed a cut-off of the left intrahepatic duct without further leakage, and the stents were removed. Imaging studies showed an atrophic left lobe, and because the liver blood test result remained normal, no further surgical intervention was considered. One patient (patient 8) developed a biliary stricture at the site of the leak, which was identified 5 months after the injury when the patient presented with abdominal discomfort, abnormal liver test results, and dilated right intrahepatic ducts on imaging studies. Although endoscopic management may have been suitable for www.giejournal.org
ERCP in pediatric bile duct injuries
this stricture, the family of the patient and the referring surgeon opted for a surgical approach as against repeated endoscopic interventions. No immediate or late complications of ERCP were observed in any of the patients.
DISCUSSION In this review, we report the results for 8 children treated endoscopically for abdominal trauma–related biliary injuries. Most of the patients initially had undergone surgery for multiple intra-abdominal organ injuries and continued to have bile leaks postoperatively. Endoscopic management of iatrogenic bile-duct injuries associated with hepatobiliary surgeries (eg, postcholecystectomy leaks and strictures) is well-established in the adult literature.1,2 The procedures include ES and/or stenting for biliary leaks as well as dilation and stenting for strictures. Non-iatrogenic injuries in the form of blunt and sharp trauma to the abdomen also can result in bile duct injuries because the liver is the most common organ affected with abdominal trauma. These injuries can be a significant source of morbidity because they are frequently associated with other organ trauma and infection. Surgical treatment, especially reoperation to manage biliary injuries, is difficult and can be associated with high morbidity including complications such as stricture formation, further leaks, and cholangitis. Endoscopic treatment of non-iatrogenic, traumatic bile duct injuries, however, may be as effective as treatment of iatrogenic injuries. Similar to iatrogenic postoperative bile leaks, case series on adults have also shown that ERCP with biliary stenting and/or ES could help in resolving bile leaks secondary to non-iatrogenic trauma.3,4 The pediatric literature on this topic is limited, with only a few case reports and one small case series.5-9 In their series of 5 patients, Castignetti et al5 demonstrated the safety and effectiveness of ERCP with biliary stenting and sphincterotomy in the noninvasive management of traumatic bile duct injuries. Their series included 5 children aged 10 years or over. Hence, the only type of endoscope used in their series was the Olympus therapeutic TJF 240 endoscope (11-mm diameter). In the current series, there were 2 cases, involving children aged 3 and 6 years, respectively, in which the PJF pediatric endoscope (7.5-mm diameter), which allows 5F accessories and stents, was used. All children in the series by Castignetti et al5 had blunt abdominal trauma with only intrahepatic bile leaks, and 60% did not require surgical intervention for multiple-organ trauma. In the present series, there was a child with a penetrating gunshot injury and another child who was run over by a school bus. The majority of the children in the current series required pre-ERCP surgical interventions for multiple-organ injuries. Despite these differences, we have shown that, irrespective of the child’s age or the nature and severity of the Volume 73, No. 4 : 2011 GASTROINTESTINAL ENDOSCOPY 825
ERCP in pediatric bile duct injuries
Ulitsky et al
Figure 2. A, Left hepatic bile leak (arrow) in patient 7 treated with sphincterotomy and placement of a 5F plastic stent. B, Hemobilia from a bleeding left hepatic artery branch was embolized 8 days later (arrowhead), and a previously placed 5F plastic biliary stent was replaced with two 5F plastic stents (block arrow). C, Follow-up ERCP showed no leakage, but there was stricturing with cut-off of the left hepatic duct (arrow).
injury, endoscopic biliary intervention is feasible, safe, and effective in closing bile leaks secondary to trauma. This study did not address the issue of whether sphincterotomy alone or in combination with stenting is required to achieve closure of intrahepatic leaks. With 8 children, the study would not have been powered enough to make this determination if the patients were randomized. In the literature on the management of iatrogenic bile leaks, the results of sphincterotomy alone, biliary stenting alone, or the combination of the two remains controversial. In our previously published series on adult patients with traumatic bile duct injuries, 7 of 8 patients underwent sphincterotomy with stenting; however, 1 patient did well with sphincterotomy alone.3 In a study of 207 adults with postcholecystectomy bile leaks, Sandha et al2 classified bile leaks as low grade (leak identified only after opacification 826 GASTROINTESTINAL ENDOSCOPY Volume 73, No. 4 : 2011
of intrahepatic ducts) or high grade (leak identified before opacification of intrahepatic ducts). Seventy-five patients with low-grade bile leaks underwent sphincterotomy alone, with successful closure of leaks in 91% of cases. The high-grade leak group had stent placement, with a 100% success rate in sealing the bile leak. In this study, there were 2 children with high-grade leaks (patients 4 and 5) and 6 with low-grade leaks. Because our first patient had a low-grade intrahepatic bile leak, we performed sphincterotomy alone in that case. Although leaks may take several days to close, in this patient the percutaneous biliary output increased after the sphincterotomy, probably secondary to postprocedure papillary edema. Hence, we brought the patient back and placed a transpapillary stent. Although no firm conclusions can be made for this patient, we elected to perform sphincterotomy with stent www.giejournal.org
Ulitsky et al
ERCP in pediatric bile duct injuries
multiple-organ trauma, as with the management of iatrogenic biliary injury, continuing bile leakage in these patients can be managed effectively and safely by using therapeutic ERCP.
placement in all subsequent patients. However, because long-term consequences of sphincterotomy in children are not known, transpapillary stenting alone may have been an attractive alternative, and this could be a subject of investigation in a prospective, multicenter, randomized study. The majority of our patients had undergone extensive surgical exploration and therapy for treatment of multiple non-biliary organ injuries before referral for ERCP. In these patients, biliary tree injuries were recognized by the bilious fluid in abdominal or chest drains or by identification of a biloma. Patients were referred for ERCP after biliary leakage continued despite the initial surgical approach. As a result of the therapeutic ERCP intervention, none of the patients required surgical repair of bile ducts to resolve bile leaks. One patient ultimately underwent surgical repair of a biliary stricture. This patient’s family preferred surgery over endoscopic therapy that may have required several procedures. The mean time for leak resolution, as defined by cessation of bilious output from abdominal or chest drains, was 12 days (range 3-20 days) after the ERCP intervention. These outcomes are similar to outcomes achieved for iatrogenic bile leaks by using ERCP interventions.3,4 In summary, as demonstrated in adults, ERCP is a safe and efficacious technique for managing biliary injuries with leaks resulting from blunt or sharp abdominal trauma in children. The use of ERCP was not associated with a higher risk of complications in children with such injuries. Although the majority of patients will undergo surgery for
REFERENCES 1. Bhattacharjya S, Puleston J, Davidson BR, et al. Outcome of early endoscopic biliary drainage in the management of bile leaks after hepatic resection. Gastrointest Endosc 2003;57:526-30. 2. Sandha GS, Bourke MJ, Haber GB, et al. Endoscopic therapy for bile leak based on a new classification: results in 207 patients. Gastrointest Endosc 2004;60:567-74. 3. Bajaj JS, Spinelli KS, Dua KS. Postoperative management of noniatrogenic traumatic bile duct injuries: role of endoscopic retrograde cholangiopancreatography. Surg Endosc 2006;20:974-7. 4. Sharma BC, Mishra SR, Kumar R, et al. Endoscopic management of bile leaks after blunt abdominal trauma. J Gastroenterol Hepatol 2009:24; 757-61. 5. Castignetti M, Houbena C, Patel S, et al. Minimally invasive management of bile leaks after blunt liver trauma in children. J Pediatr Surg 2006;41: 1539-44. 6. Keil R, Snajdauf J, Stuj J, et al. Nonoperative therapy of the posttraumatic biliary fistula in adolescents. Eur J Pediatr Surg 2001;11:274-6. 7. Sharpe RS, Nance ML, Stafford PW. Nonoperative management of blunt extrahepatic biliary duct transection in the pediatric patient: case report and review of the literature. J Pediatr Surg 2002;37:1612-6. 8. Church NG, May G, Sigalet DL. A minimally invasive approach to bile duct injury after blunt liver trauma in pediatric patients. J Pediatr Surg 2002; 37:773-5. 9. Roche B, Mentha G, Bugmann P, et al. Intrahepatic biliary lesions following blunt liver trauma in children: is nonoperative management or conservative operative treatment always safe? Eur J Pediatr Surg 1993;3: 209-12.
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Volume 73, No. 4 : 2011 GASTROINTESTINAL ENDOSCOPY 827