Hepatic Artery Aneurysm Rupture After Double‐Balloon Endoscopy in Biliary Atresia

A 37-year-old man with biliary atresia, for which he underwent a Kasai procedure (Roux-en-Y reconstruction) at 60 days of age, was admitted for cholangitis. He received antibiotics and, due to multiple intrahepatic gallstones (Figure 1), was scheduled for double-balloon endoscopic retrograde cholangiography and lithotripsy. However, severe intraperitoneal adhesions impaired scope maneuverability, limiting access to the portoenterostomy. Moderate esophageal and jejunal varices were present. Contrast medium was injected from the jejunum, which served as the afferent loop, revealing multiple stones in the jejunum and liver (Figure 2). The stones could not be removed endoscopically. Dynamic computed tomography (CT) of a 37-year-old patient with biliary atresia post-Kasai procedure with Roux-en-Y reconstruction (equilibrium phase, coronal section) was performed prior to the attempt of double-balloon endoscopic retrograde cholangiography. Multiple intrahepatic stones, each up to 1 cm in diameter and considered to be the cause of cholangitis, were identified (white arrowheads). Double-balloon endoscopic retrograde cholangiography was attempted for the first time for cholangitis due to intrahepatic bile stones. Due to intraperitoneal adhesion, as felt by the endoscopist during the procedure, the scope could only be inserted up to the middle of the jejunum, which served as the afferent loop (black arrow) and did not reach the portoenterostomy. Since biliary cannulation was impossible, iodine contrast agent was injected into the jejunum to evaluate the biliary tract as precisely as possible. Multiple stones in the liver (white arrowheads) and jejunum (black arrowheads) were present, while stenosis at the portoenterostomy was not evident on fluoroscopic images (white arrows). Three days post-endoscopy, the patient experienced abdominal pain and bloody stool. His hemoglobin level dropped from 10.1 to 7.9 g/dL. Abdominal imaging revealed a hepatic artery aneurysm (HAA) and blood in the intestine which had not been present before endoscopy (Figure 3), leading to emergency angiography. Trans-arterial embolization with 20% n-butyl-2-cyanoacrylate was performed (Figure 4), and the patient's recovery was favorable. Three days after performing double-balloon endoscopy, the patient exhibited bloody stool, abdominal pain, and progression of anemia, suggesting the possibility of gastrointestinal bleeding. He underwent dynamic computed tomography (CT) again to identify the bleeding source. (a) The arterial phase of dynamic CT (axial section) revealed an 18 mm × 16 mm-sized enhancement in the right lobe of the liver, leading to the diagnosis of a hepatic artery aneurysm (HAA) (white arrow). (b) Dynamic CT performed 2 days before the endoscopy showed no evidence of HAA. Therefore, it was concluded that the HAA developed and ruptured shortly after the procedure. Emergency angiography for hemostasis of the hepatic artery aneurysm was performed. Angiography of the right hepatic artery revealed the aneurysm (white arrow). A feeding vessel branched from A8a (white arrowheads). After determining that this vessel was the cause of the bleeding, embolization with 20% n-butyl-2-cyanoacrylate was subsequently performed. Post-embolization angiography of the hepatic artery showed that the hepatic artery aneurysm was sufficiently embolized. HAAs are rare and occur for various reasons [1], including liver surgery [2] and biliary stent deployment [3-5]. This case is unique, as it is the first reported case where an HAA formed and ruptured shortly after endoscopy without any biliary intervention, long after abdominal surgery. Because of severe intraperitoneal adhesions, tension by scope manipulation might have been transmitted to the portoenterostomy, potentially causing damage to the hepatic artery. When performing endoscopy on patients with post-surgical adhesion, extremely gentle endoscopic manipulation is crucial. If bloody stool occurs post-procedure, a ruptured HAA should be considered as one of the potential diagnoses. The authors have nothing to report. Written informed consent was obtained from the patient presented in this document. The authors declare no conflicts of interest. The authors have nothing to report. The authors have nothing to report.