Life-threatening hemobilia caused by hepatic pseudoaneurysm after T-tube choledochostomy: report of a case

Background Hemobilia is a rare but lethal biliary tract complication. There are several causes of hemobilia which might be classified as traumatic or nontraumatic. Hemobilia caused by pseudoaneurysm might result from hepatobiliary surgery or percutaneous interventional hepatobiliary procedures. However, to our knowledge, there are no previous reports pertaining to hemobilia caused by hepatic pseudoaneurysm after T-tube choledochostomy. Case presentation A 65-year-old male was admitted to our hospital because of acute calculous cholecystitis and cholangitis. He underwent cholecystectomy, choledocholithotomy via a right upper quadrant laparotomy and a temporary T-tube choledochostomy was created. However, on the 19th day after operation, he suffered from sudden onset of hematemesis and massive fresh blood drainage from the T-tube choledochostomy. Imaging studies confirmed the diagnosis of pseudoaneurysm associated hemobilia. The probable association of T-tube choledochostomy with pseudoaneurysm and hemobilia is also demonstrated. He underwent emergent selective microcoils emobolization to occlude the feeding artery of the pseudoaneurysm. Conclusions Pseudoaneurysm associated hemobilia may occur after T-tube choledochostomy. This case also highlights the importance that hemobilia should be highly suspected in a patient presenting with jaundice, right upper quadrant abdominal pain and upper gastrointestinal bleeding after liver or biliary surgery.


Case Presentation
A 65-year-old male was admitted to our hospital because of right upper quadrant abdominal pain, jaundice and fever for 2 days. He denied history of any systemic disease. He had no history of previous abdominal surgery. At initial presentation, his body temperature was 38.6°C, blood pressure was 142/82 mmHg, pulse rate was 92/ min, and respiratory rate was 22/min. Physical examination revealed jaundice, and tenderness with light palpation at the right upper quadrant of abdomen. Murphy's sign was positive. There was no palpable mass and Courvoisier's sign was negative. The bowel sound was normal in peristalsis and no abdominal bruit was found on auscultation.
Because acute calculous cholecystitis and cholangitis were impressed on clinical grounds, he underwent cholecystectomy and choledocholithotomy via a right upper quadrant laparotomy. At operation, an inflamed gallbladder with fibrin coating on the edematous wall was found. The Calot's triangle showed dense adhesion and the CBD was distended. The CBD was explored to remove sandy stones, debris and sticky bile. A temporary T-tube choledochostomy was created and Penrose drains were set in the Morison's pouch.
The patient recovered well after the operation without fever episodes or jaundice. He was discharged on the 14 th postoperative days. However, on the 19 th day after operation, he suffered from sudden onset of hematemesis and massive fresh blood drainage from the T-tube choledochostomy. Hypovolemic shock developed thereafter. He was sent to emergent department of our institution. Laboratory examination revealed a decreased hemoglobin level of 7.0 g/dL and an increased total bilirubin level of 2.1 mg/dL. Because hemobilia was highly suspected a contrast enhanced computed tomography (CT) of the abdomen was arranged emergently, revealing a round and hyperdense lesion abutting the T-tube choledochostomy (Figure 1). The reconstructive CT image further demonstrated a saccular mass protruding from the proper hepatic artery (Figure 2). The angiography disclosed a contrast-filled lesion arising from proper hepatic artery, which suggested a pseudoaneurysm formation (Figure 3). The contrast drained into the T-tube choledochostomy directly (Figure 4), indicating that pseudoaneurysm was the cause of hemobilia in our patient. He underwent emergent selective microcoils embolization to occlude the feeding artery of the pseudoaneurysm and the bleeding ceased thereafter ( Figure 5). He recovered unevent-fully with normal liver profile after a three-month period of follow up.
As for the etiological cause of pseudoaneurysm formation in our case, we propose that mechanical compression of the T-tube on the bile duct mucosa and adjacent proper hepatic artery might have caused erosion of the blood vessel, and induced pseudoaneurysm formation. There are evidences that might support our proposal. Firstly, anatomically speaking, the common bile duct runs along with the proper hepatic artery within the hepatoduodenal ligament. Secondly, the contrast-filled hyperdense pseudoaneurysm found at abdominal CT was just close to the T-tube. The angiographic features further disclosed that the lunar pseudoaneurysm was compressed by the T-tube and the contrast flew directly from the proper hepatic artery into the T-tube.
The diagnosis of hemobilia can be confirmed by endoscopy examination or endoscopic retrograde cholangiopancreatography to directly observe bleeding from the ampulla of Vater [12]. Doppler ultrasound can be used to detect the pulsatile mass with blood flow into the biliary tracts [24]. Tumors, abscesses, and the location of aneurysms and pseudoaneurysms can be identified by abdominal CT [25]. Furthermore, contrast-enhanced magnetic resonance imaging can be used to demonstrate the correlation of blood flow with bile ducts to support the diagnosis of hemobilia [26]. However, transarterial angiography should be considered as the imaging modality of choice, because it can be used to detect and embolize the bleeders [2,9,22,27,28]. Surgical ligation, excision of aneurysm and pseudoaneurysm or partial hepatectomy is only conserved for failed embolization or tumor-related hemobilia [2,22]. Furthermore, recent articles suggest that minimally invasive procedures were offered for treatment of hemobilia, especially for tumor associated hemobilia [18,19,22].
In conclusion, pseudoaneurym associated hemobilia may occur after T-tube choledochostomy. This case also highlights the importance that hemobilia should be highly suspected in a patient presenting with jaundice,   right upper quadrant abdominal pain and UGI bleeding after liver and biliary surgery. Once a diagnosis is established, angiographic intervention should be performed without delay because it is a life-threatening situation.

Consent
Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.