Abstract

INTRODUCTION: Pseudoaneurysm of the cystic artery is a rare cause of hemobilia and often derives from the inflammatory changes in cholecystitis. Knowledge regarding this topic is limited with rare occurrence reported in the literature. This may be the first reported case of a cystic artery pseudoaneurysm causing biliary obstruction in a pediatric patient. This case emphasizes the importance of considering hemobilia whenever gastrointestinal bleeding is associated with cholestatic liver injury as well as including cystic artery aneurysm in the differential diagnosis of hemobilia. CASE DESCRIPTION/METHODS: A 16-year-old obese male scheduled for elective cholecystectomy for symptomatic cholelithiasis was admitted for worsening abdominal pain associated with nausea and vomiting. After admission, patient remained afebrile with resolving symptoms, but rising cholestatic markers. MRCP showed Common Bile Duct (CBD) dilation to 1 cm but without CBD stones. EUS was performed to evaluate for choledocholithiasis but was limited by the unexpected finding of fresh blood in the duodenum. CBD dilation to 8 mm was seen but was without evidence of CBD stones. Cholestatic injury worsened, prompting ERCP as distal CBD stones were still the leading concern. Upon examination prior to cannulation, passage of blood and blood clots was seen from the native ampulla. Cholangiogram showed multiple filling defects within the CBD suggestive of blood clots, sphincterotomy was performed and multiple sweeps with the balloon catheter removed only blood clots with no stones seen. CT angiography was performed and found a focal arterial density collection in the posteromedial gallbladder wall, most likely due to a pseudoaneurysm of the cystic artery. Interventional Radiology performed coil embolization of the cystic artery prior to cholecystectomy. Laparoscopic cholecystectomy was converted to open laparotomy after the discovery of extensive pericholecystic adhesions to the gallbladder as well as a cholecystocolonic fistula. The fistula was repaired and the gallbladder was removed with ligation of the cystic artery just distal to the coils. Symptoms resolved and the patient's laboratory values normalized. DISCUSSION: A rare case of cholestatic liver injury and hemobilia secondary to cystic artery pseudoaneurysm in a pediatric patient is reported. As demonstrated in this report, immediate angiography and embolization of the involved artery followed by cholecystectomy may be the optimal treatment regardless of patient age.

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