Emergency cholecystectomy and hepatic arterial repair in a patient presenting with haemobilia and massive gastrointestinal haemorrhage due to a spontaneous cystic artery gallbladder fistula masquerading as a pseudoaneurysm

Abstract

BackgroundHaemobilia usually occurs secondary to accidental or iatrogenic hepatobiliary trauma. It can occasionally present with cataclysmal upper gastrointestinal haemorrhage posing as a life threatening emergency. Haemobilia can very rarely be a complication of acute cholecystitis. Here we report a case of haemobilia manifesting as massive gastrointestinal haemorrhage in a patient without any prior history of biliary surgery or intervention and present a brief review of literature.Case presentationA 22 year old male admitted with history suggestive of acute cholecystitis subsequently developed waxing waning jaundice and recurrent episodes of upper gastrointestinal bleed. Endoscopy showed an ulcer in the first part of duodenum with a clot, no active bleed was visible. Angiography was suggestive of a ruptured pseudoaneurysm in the vicinity of the right hepatic artery probably originating from the cystic artery. Coil embolization was tried but the coil dislodged into the right branch of hepatic artery distal to the site of pseudoaneurysm. Review of angiographic video in light of operative findings demonstrated a fistulous communication between cystic artery and gallbladder as the cause, a simultaneous cholecystoduodenal fistula was also noted. Retrograde cholecystectomy, closure of cholecystoduodenal fistula and right hepatic arteriotomy with retrieval of the endo-coil and hepatic arterial repair was performed.ConclusionFistula between the cystic artery and gallbladder has been commonly reported to occur after laparoscopic cholecystectomy. Spontaneous fistulous communication, i.e. in the absence of any prior trauma or intervention, between cystic artery and gallbladder is rare with very few reports in literature. Aetiopathogenesis of the disease, in the context of current literature is reviewed. The diagnostic dilemma posed by the confounding finding of an ulcer in the duodenum, the iconic video angiographic depiction as also the therapeutic challenge of a failed embolization with consequent microcoil migration and primary hepatic arterial repair in the emergency situation is discussed.

Spontaneous right hepatic artery branch gallbladder fistula revealed by haemobilia and upper cataclysmic gastrointestinal bleeding

“…In the literature, few cases of spontaneous fistula between cystic artery and gall bladder are reported 8. To our knowledge, no similar case of spontaneous right hepatic artery fistula into the gall bladder has been reported to date.…”

Acute hemobilia from a pseudoaneurysm of the cystic artery arising from the left hepatic artery: Case report and literature review

“…It has a high percentage of success reaching hemostasis in 75%–100% of patients with hemobilia [20] with a reported complication of less than 2% [21].…”

Transarterial catheter embolisation for an unusual cause of upper gastrointestinal haemorrhage

“…The majority (85% cases4) of reported cystic artery pseudoaneurysms have been attributed to traumatic or postsurgical causes 56 Only 21 cases of cystic artery pseudoaneurysm formation relating to calculous cholecystitis have been reported in the medical literature before 2013,7 with a further four cases published within the last year 8–11…”

“…The classical signs and symptoms of haemobilia consist of biliary colic, obstructive liver function tests and upper gastrointestinal bleeding—the so-called ‘Quinke's Triad’ 7. Nevertheless, the diagnosis is rarely clear-cut as these three modes of presentation occur in only 32–40% of cases and rarely all at the same time 17–21…”

“…Complications include gallbladder ischaemia21 and perforation, coil migration into bile ducts and recurrent haemorrhage 2932 In a case report by Priya et al ,7 migration of a microcoil resulted in recurrent haemorrhage and emergency surgery was necessary to control the bleeding.…”