Pseudoaneurysm of anomalous cystic artery due to calculous cholecystitis

Abstract

Pseudoaneurysm of the cystic artery is a rare cause of haemobilia resulting from either an inflammatory process in the abdomen or abdominal trauma. We report a case of a patient with chronic calculous cholecystitis associated with a pseudoaneurysm arising from an anomalous cystic artery who presented with haemobilia. The patient was managed successfully with multimodality treatment that included angioembolisation of the pseudoaneurysm and stenting of the common bile duct to relieve jaundice followed by elective open cholecystectomy.

Background

Gastrointestinal bleeding from the biliary tree (haemobilia) is an uncommon event. Previously, the most common cause of this disease was traumatic hepatobiliary injury, however, iatrogenic trauma caused by percutaneous and endoscopic hepatobiliary procedures is now the cause in two-third of haemobilia cases.¹ Other causes are gallstones, inflammatory diseases, vascular malformations and neoplastic disease.¹ These aneurysms can be either congenital or acquired. Pseudoaneurysms due to cholecystitis are very rare. We report a rare case of haemobilia due to a pseudoaneurysm of an anomalous cystic artery secondary to chronic calculus cholecystitis with Mirrizi's syndrome.

Case presentation

A 55-year-old man presented to the emergency department of our tertiary referral hospital with a 4-month history of repeated episodes of jaundice, right upper abdominal pain and malena. One year earlier the patient had been diagnosed with gall stones. On clinical examination the patient was pale and icteric, with no significant abdominal signs. Per rectal examination revealed malena.

Investigations

Blood investigations showed anaemia with direct hyperbilirubinaemia. Abdominal ultrasound revealed gallstones with mild central intrahepatic biliary radicle dilation.

The contrast-enhanced CT of the abdomen showed a pseudoaneurysm arising from the medial aspect of the gallbladder wall with a mass suspected to be a stone in the gallbladder (figure 1).

Figure 1.

CT angiography showing cystic artery pseudoaneurysm arising from the gallbladder wall.

MR cholangiography revealed a pseudoaneurysm of the cystic artery (figure 2A,B) with blood clots, a stone within the gallbladder and type I Mirrizi’s syndrome (figure 2C). The cystic artery bearing the pseudoaneurysm was found to originate directly from the common hepatic artery on selective hepatic catheterisation (figure 3A). Early branching of the left hepatic artery from the common hepatic artery was also seen (figure 3A).

Figure 2.

MR angiography of the abdomen (A) cystic artery pseudoaneurysm on axial images, (B) cystic artery pseudoaneurysm on sagittal images, (C) MR cholangiopancreatography showing mid-common bile duct narrowing with intrahepatic biliary radical dilatations.

Figure 3.

Digital subtraction angiography (A) before embolisation showing anomalous cystic artery with pseudoaneurysm at its tip, (B) after embolisation.

Differential diagnosis

• Cholelithiasis with choledocholithiasis

• Mirrizi's syndrome

• Hepatic artery pseudoaneurysm

Treatment

Super selective catheterisation with microcoil embolisation of the anomalous cystic artery was performed resulting in occlusion of the pseudoaneurysm (figure 3B).

Endoscopic retrograde cholangiography (ERC) was performed for direct hyperbilirubinaemia. On ERC, there was passage of blood clots from the ampulla of Vater with extrinsic compression on the mid-portion of the common bile duct (CBD) without any filling defects. CBD stenting was performed to relieve jaundice.

Six days later the patient underwent elective open subtotal cholecystectomy with under-running of the cystic artery pseudoaneurysm. Intraoperatively, the gallbladder was thickened, distended and intrahepatic, and contained pus; it also contained a single large stone. There were dense adhesions in Calot's triangle. An ovoid vascular lesion was found projecting into the lumen of the gallbladder from the posterosuperior wall of its body (figure 4). The gallbladder wall around it was left behind as it was densely adhered to the liver.

Figure 4.

Intraoperative photograph of pseudoaneurysm during open cholecystectomy.

Outcome and follow-up

The patient had an uneventful postoperative recovery with regression of symptoms including jaundice and malena. Histopathology revealed acute on chronic cholecystitis. On follow-up after 2 years he was operated for an inguinal hernia and since then he has been asymptomatic.

Discussion

The association of upper gastrointestinal bleeding with signs of biliary disorders should suggest haemobilia. Haemobilia is usually associated with Quincke's triad of biliary colic (occurring in 70% of patients), jaundice (exhibited in 60% of cases) and gastrointestinal bleeding, which is present in all patients. The complete classic triad is found in about 22% of patients¹ with acute bleeding first causing biliary colic followed by haematemesis or malena, which in turn leads to pain relief.

A cystic artery pseudoaneurysm due to an inflammatory process is a rare cause of haemobilia; an extensive search identified only 58 published cases (table 1).

Table 1.

Summary of cases of cystic artery pseudoaneurysm secondary to inflammation reported in literature

Serial number	Author	Year	Journal	Aetiology	Presentation	Management
1	Glaysher et al	2014	International Journal of Surgery Case Reports	Calculous cholecystitis with cholecystoenteric fistula	Jaundice+pain+malena	Open subtotal cholecystectomy
2	Suzuki et al	2013	Clinical Journal of Gastroenterology	Acute calculous cholecystitis	Epigastric pain+jaundice	Open cholecystectomy
3	Nana et al	2013	International Journal of Surgery Case Reports	Acute cholecystitis	Quinke triad	TAE+lap cholecystectomy
				Acute cholecystitis	Haemobilia+pain	TAE
4	Fung et al	2013	Scottish Medical Journal	Acute cholecystitis	Haemobilia	–
5	Dewachter et al	2012	Journal Belge de Radiologie - Belgisch Tijdschrift voor Radiologi	Acute calculous cholecystitis	Epigastric pain+nausea	Lap cholecystectomy+resection of pseudoaneurysm
6	Chong et al	2012	Canadian Association of Radiologists Journal	–	Fever+epigastric pain+vomiting	–
7	Komatsu et al	2011	Journal of Medical Cases	Acute calculous cholecystitis	Epigastic pain+haematemesis+malena	TAE
8	Anand et al	2011	Annals of Gastroenterology	Idiopathic	RUQ pain+haematemesis+malena	Cholecystectomy
9	Nkwam et al	2010	Journal of Surgical Case Reports	Acute calculous cholecystitis	RHC pain	TAE
10	Desai et al	2010	Radiology Case	Chronic calculous cholecystitis	Epigastic pain+malena	TAE
11	Ahmed et al	2010	The British Journal of Radiology	Xanthogranulomatous cholecystitis	RHC pain+lump	TAE+open cholecystectomy
12	Leung et al	2010	Hong Kong Medical Journal	Acute cholecystitis	Haematochezia	TAE
13	Hague et al	2010	CardioVascular and Interventional Radiology	Acute calculous cholecystitis	RHC pain	TAE
				Acute calculous cholecystitis	RHC pain	TAE
				–	Epigastric pain+malena	TAE
14	Siddique et al	2011	BMJ Case Reports	Acute xanthogranulomatous cholecystitis	Epigastric pain+fever	TAE+elective open cholecystectomy
15	Mullen et al	2009	CardioVascular and Interventional Radiology	Acute calculous cholecystitis	RHC pain+malena	TAE
				Acute calculous cholecystitis	RHC pain	TAE
16	Sousa et al	2009	Gastroentérologie Clinique et Biologique	Acute calculous cholecystitis	Epigastric pain+haematemesis	Cholecystectomy
17	Radouane et al	2008	Journal of Radiology	Calculous cholecystitis	Not known
18	Machida et al	2008	Radiation Medicine	Acute calculus cholecystitis	RUQ pain	Cholecystectomy
19	Al’ Aref et al	2008	Hepatobiliary & Pancreatic Diseases International	Idiopathic	RUQ pain+malena	TAE
20	Shimada et al	2008	Digestive Surgery	Xanthogranulomatous cholecystitis	Jaundice	TAE—extended right hepatectomy
21	Ghoz et al	2007	Hepatobiliary & Pancreatic Diseases International	Acute on chronic cholecystitis	Abdominal pain+haematemesis	TAE+cholecystectomy
22	Akatsu et al	2007	Surgery Today	Acute cholecystitis	RUQ pain+jaundice	Cholecystectomy
23	Saluja et al	2007	BMC Gastroenterology	Acute calculus cholecystitis	Haematemesis+Malena	TAE+partial cholecystectomy
24	Chun-Jung Lin et al	2007	Chinese Journal of Radiology	Acalculous cholecystitis	Joundice+abdominal pain	TAE+cholecystectomy
25	Pérez-Castrillón et al	2006	Endoscopy	Acute cholecystitis	Abdominal pain+anaemia	TAE
26	Lee	2006	Clinical Radiology	Chronic cholecystitis	Lower GI bleed	Not known
27	Sibulesky et al	2006	American Journal of Surgery	Calculous cholecystitis	RHC pain+malena	Open cholecystectomy
28	Joyce et al	2006	Irish Journal of Medical Sciences	Acute on chronic cholecystitis	Haemobilia	Cholecystectomy
29	Oueslati et al	2005	Journal de Chirurgie	Idiopathic	Haemobilia	Cholecystectomy
30	Morioka et al	2004	Journal of Gastroenterology & Hepatology	Chronic calculus cholecystitis	Haemobilia	Cholecystectomy
31	Gutierrez et al	2004	American Journal of Surgery	Acute calculus cholecystitis	RHC pain+malena	TAE failed f/b open cholecystectomy
32	Hiroshi et al	2002	Journal of Biliary Tract Pancreas		Dyspnoea+malena	TAE
33	Maeda et al	2002	Journal of Hepato-Biliary-Pancreatic Surgery	Calculous cholecystitis	Epigastric pain+jaundice	TAE+elective open cholecystectomy
34	AA Palejwala et al	2000	CME Journal Gastroenterology, Hepatology and Nutrition	Acute cholecystits	RUQ pain+haematemesis+jaundice	Cholecystectomy
35	Delgadillo et al	1999	Journal of Vascular & Interventional Radiology	Acute pancreatitis	Haemobilia	TAE
36	dePerrot et al	1999	The British Journal of Surgery	Pancreatitis	Epigastric pain	Surgery
37	Kaman et al	1998	American Journal of Gastroenterology	Acute calculous cholecystitis	Haemobilia	Cholecystectomy with ligation of pseudoaneurysm
38	Kirchgatterer et al	1998	Wiener Klinische Wochenschrift	Chronic calculous cholecystitis	Haemobilia	Cholecystectomy
39	England et al	1998	Clinical Radiology	Acute calculous cholecystitis	Haemobilia	Open cholecystectomy f/b TAE
40	Miura et al	1998	Nippon Shokakibyo Gakkai Zasshi	–	Haemobilia	–
41	Ritz et al	1997	Journal de Chirurgie	Acute calculous cholecystitis	Haemobilia	Cholecystectomy
42	Matsuba et al	1996	Japanese Journal of Gastroenterology Surgery	Cholecystitis	RUQ pain	Cholecystectomy
43	Nakajima et al	1996	Journal of Gastroenterology	Acute calculous cholecystitis	Haemobilia	Cholecystectomy
44	Barba et al	1994	Canadian Journal of Surgery	Cholecystitis	Haemobilia	Cholecystectomy with ligation of pseudoaneurysm
45	Read et al	1991	ANZ Journal of Surgery	Cholecystitis	Haemobilia	Cholecystectomy
46	Strickland et al	1991	CardioVascular and Interventional Radiology	Calculous cholecystitis	Haemobilia	–
47	Smague et al	1990	Journal de Chirurgie	Acute cholecystits	Upper abdominal pain+haematemesis	Cholecystectomy
48	Read et al	1990	ANZ Journal of Surgery	Chronic calculous cholecystitis	Haemobilia	Cholecystectomy
49	Wu et al	1988	Acta Chirurgica Scandinavica	Acute cholecystitis	–	Surgery
50	Rhee JW et al	1987	New York State Journal of Medicine	Acute calculous cholecystitis	RUQ pain+fever	Cholecystectomy
51	Reddy et al	1983	Southern Medical Journal	Cholecystitis	Haemobilia	Cholecystectomy
52	Glazer et al	1980	Journal of Radiology	Postcholecystectomy	UGI bleed	–
53	Hakami et al	1976	American Journal of Proctology	? Cholecystitis	Haemobilia	Cholecystectomy
54	Devin et al	1971	Journal de Chirurgie	Acute pancreatitis	Epigastric pain, jaundice, malena	Partial cholecystectomy

?, suspected; f/b, followed by; GI, gastrointestinal; RHC, right hypochondrium; RUQ, right upper quadrant; TAE, transarterial embolisation; UGI, upper GI.

The exact cause of association between pseudoaneurysms and inflammation is not clear but it has been hypothesised that the inflammatory process leads to ulceration of the gallbladder wall and partial erosion of the elastic and muscular components of the vascular wall, leading to the development and rupture of the pseudoaneurysm.² Despite cholecystitis being common, the rarity of cystic artery pseudoaneurysms can be explained by early thrombosis of the cystic artery due to inflammation.³

Cystic artery pseudoaneurysms can rupture in the biliary tree⁴ or rarely into the peritoneum.⁵ A high index of suspicion is required for diagnosis. Abdominal colour Doppler ultrasound can be an effective initial diagnostic tool but lacks high sensitivity.⁴ Endoscopy is helpful in diagnosing haemobilia in a patient with upper gastrointestinal bleeding. MRI and CT scan can identify the underlying pathology such as stones, cholecystitis, neoplasms and vascular abnormalities. However, selective hepatic angiography is the technique of choice in the management of haemobilia of any cause and for suspected pseudoaneurysms in particular, due to its high diagnostic accuracy and therapeutic potential.^{1 6} It is useful especially in elderly, haemodynamically unstable patients unfit for surgery, in stopping the active bleeding. Nevertheless, angiography has certain diagnostic limitations for the variable flow rate and intermittent bleeding, and also when there are hepatic artery abnormalities or when there has been previous manipulation. Despite there being reports of gallbladder infarction after hepatic artery embolisation with occlusion of the cystic artery, there has been no report of gallbladder gangrene, to date, after embolisation of the cystic artery pseudoaneurysm, probably due to collateral blood supply from the epicholedochal artery.⁷

In our patient, the cystic and left hepatic arteries were arising from the common hepatic artery. These anomalies have been described in the literature but are very uncommon.^{8 9}

Haemobilia is known to cause biliary obstruction due to blood clots leading to jaundice, as seen in our case, which can be relieved by endoscopic drainage with stenting or nasobiliary drainage.¹⁰

Maeda et al⁵ were the first to report successful management of a cystic artery aneurysm by a combined approach: embolisation of the cystic artery to stabilise the patient, followed by cholecystectomy at a later, safer time. It seems a logical combination as the cause of inflammation, the stone, needs to be treated as well. Our case was also managed similarly with success.

Learning points.

• In patients with gallstone disease, presence of malena should raise suspicion of a pseudoaneurysm.

• A cystic artery pseudoaneurysm can be successfully managed by angioembolisation and open total/subtotal cholecystectomy.

• Patients with a cystic artery pseudoaneurysm not fit for surgical intervention can be managed by embolisation alone.