Successful Treatment of a Pseudoaneurysm of the Cystic Artery with Microcoil ~mbolizationl Xavier Delgadillo, MD Thierry Berney, MD, MS Marc de Perrot, MD Dominique Didier, MD Philippe Morel, MD
Index terms: Cystic artery, aneurysm * Cystic artery, therapeutic blockade Gastrointestinal tract, bleeding
.
JVIR 1999; 10:789-792
PSEUDOANEURYSMS of visceral arteries are uncommon but wellcharacterized vascular abnormalities, usually provoked by intraabdominal inflammatory processes such as pancreatitis or cholecystitis, or by surgical trauma (1). However, pseudoaneurysms of the cystic artery are rare. They complicate cholecystitis or cholecystectomy, and manifest as hemobilia as they rupture into the biliary tree (2). The advent of transcatheter embolization techniques has begun to allow minimally invasive treatment of these life-threatening complications (1,3,4). Transcatheter embolization can be performed using several types of material, such as synthetic occlusive emulsions, gelatin sponges or other particles, or metallic microcoils. Microcoils are small metallic helical particles, made of stainlesssteel, platinum, or tungsten. Superselective catheterization of an artery and release of microcoils causes the vessel to thrombose and allows control of bleeding (5).
CASE REPORT
From the Clinic of Digestive Surgery and the D ~ (X,D,,T,B,, M,dP,, ment of Radiology (D.D.),Geneva University Hospital, 1211 Geneva 14, Geneva, Received September 3, revision requested October 13; revision received November 12; accepted N ~ ber 13. Address correspondence to T.B., Diabetes Research Institute, 1450 NW loth Ave, Miami, FL 33136.
o SCVIR,
1999
A 28-year-old, HIV-positive man receiving stavudine, saquinavir, and zidovudine triple therapy was admitted to the emergency ward for a 3-day history of epigastric and right upper ~ ~ quadrant ~ ~ pain, - with nausea and alimentary vomiting. He had no history of opportunistic infection. On examination, he had a low-grade fever ~ ~(37.g°C) ~ -and epigastric tenderneSS without guarding. Laboratory findings showed a disturbance of the liver and pancreas blood tests (AST, 310 IUL; ALT, 209 IUL; al-
kaline phosphatase, 712 IUL; gamma-GT, 2,076 IUL; total bilirubin, 76 mmolJL; direct bilirubin, 59 mmoVL; amylase, 350 IUL; and lipase, 1,167 IUL). His white blood cell count was 6,900/mm3, without left shift, and his hemoglobin was 12.2 g/dL. Initial abdominal ultrasonography (US) revealed a heterogeneous thickening of the gallbladder wall and a 3 x 2-cm hyperechogenic mass at the porta hepatis. Six hours after admission, the patient had a massive episode of hematemesis, his hemoglobin dropped to 6.4 g/dL, but his hemodynamics remained stable. Three units of packed red blood cells were transfused. After washing of large amounts of blood collected in the stomach and duodenum, emergency endoscopic examination showed a blood clot situated on a thickened fold of the second duodenal mucosa. There was no active bleeding. This was interpreted as a duodenal peptic ulcer, and the examiner injected its margins with adrenaline to prevent bleeding recurrence. Control endoscopy 24 hours later showed that bleeding had stopped, but because of the unusual localization of the suspected ulcer, an abdominal computed tomographic (CT) scan was obtained. It revealed a hematoma at the porta hepatis and within the gallbladder, surrounding a 1.5-cm, nodular image enhanced by contrast material injection (Fig 1). A diagnosis of arterial pseudoaneurysm was made. Signs of global edematous pancreatitis were present. Selective arteriography of the right hepatic artery was immediately performed and revealed a
789
790
Embolization of Cystic Artery Pseudoaneurysm
June 1999 JVIR
Figure 1. Abdominal CT scan showing a hematoma a t the porta hepatis and within the gallbladder, surrounding a 1.5-cm nodular image enhanced by contrast material injection (arrow).
filling of the branch to the gallbladder (Fig 2b). Immediate follow-up was marked by a rapid disappearance of symptoms and a return of the liver and lsancreas blood tests to normal levels. On control arteriography performed a few days later, the pseudoaneurysmal lesion had disappeared and the gallbladder was normally vascularized. Abdominal US and CT examinations showed a complete disappearance of radiologic signs of pancreatitis but did not permit satisfactory analysis of the gallbladder, which appeared globally hyperechogenic and was interpreted a s scleroatrophic. Gallstones could not be absolutely demonstrated or ruled out. The patient was discharged on day 18 after admission. A cholecystectomy was envisaged, but was declined by the patient for financial reasons. The patient was seen once as a n outpatient 5 months after the bleeding episode, and was free of recurrence a t that time. The patient was lost to further follow-up shortly thereafter.
I DISCUSSION
Figure 2. Digital subtraction angiography of the right hepatic artery revealing a pseudoaneurysm (arrow) of a branch of the cystic artery bordering the gallbladder with extravasation of contrast material (a).After hyperselective catheterization of this branch and embolization with four microcoils, absence of opacification of the aneurysmal arterial branch but normal filling of the branch to the gallbladder (arrow) can be seen (b).
pseudoaneurysm of a branch of the cystic artery bordering the gallbladder, with extravasation of contrast material (Fig 2a). Hyperselective catheterization of this branch with a 5-F catheter allowed embolization
using four stainless-steel microcoils (1mm and 1.5 mm diameter) (Hilal; Cook, Bloomington, IN). Subsequent injection of the cystic artery showed absence of opacification of the aneurysmal arterial branch, but normal
The cystic artery is a rare localization for pseudoaneurysms; only 12 cases have been reported so far in the literature (1-4,6-13). All published cases occurred either after cholecystectomy or during a n episode of acute cholecystitis. It seems that more cases have been observed since the advent of laparoscopic cholecystectomy (14). Characteristics of these 12 published cases, and of three additional cases of postcholecystectomy pseudoaneurysms of a branch of the right hepatic artery (14), which is likely to be the stump of the cystic artery, are shown in the Table. Embolization of the cystic artery has been attempted in seven cases with various materials, including Ethibloc (Ethicon, Somerville, NJ), an emulsion that solidifies after injection to provide a cast of the aneurysm (141, Oxycel (Paske-Davis, Morris Plains, NJ) pledgets (12), Gelfoam (Upjohn, Kalamazoo, MI) sponges (11, and microcoils (3,4). To our knowledge,
Delgadillo et a1
791
Volume 10 Number 6
Characteristics of 15 Published Cases of Bleeding Pseudoaneurysms of the Cystic Artery* Etiology
Patients
TCE
Material?
No.
Outcome
Postcholecystectomy~
8
6
Cholecystitis
7
1
Occlusion emulsion Hemostatic sponges Microcoils Microcoils
3 2 1 1
Embolization success Complications$ Surgical treatment1 Embolization failure Surgical treatment¶
6 2 2 1 7
Note.-TCE = transcatheter embolization. #: Including three cases of postcholecystectomy pseudoaneurysms of a branch of the right hepatic artery, most likely the stump of the cystic artery. f Material used for transcatheter embolization. f Six patients after laparoscopic cholecystectomy, two patients after open cholecystectomy. $ Complications of transcatheter embolization: one subphrenic hematoma; one nonselective embolization of the common hepatic artery. 1 Cholecystectomy and resection of the pseudoaneurysm.
this is the second reported case of successful microcoil embolization of the cystic artery. The etiology of pseudoaneurysms of the hepatic artery and its branches is manifold, and includes blunt abdominal trauma, previous surgery of the biliary tract or liver biopsy, cholecystitis, and pancreatitis (12). All of these stimuli act through partial erosion of the serosa and the elastic and muscular components of the arterial wall to give rise to the pseudoaneurysm. Its natural history is that of progressive enlargement, eventually leading to rupture (101, a phenomenon enhanced by the ongoing inflammatory process in the case of cholecystitis or pancreatitis. On the other hand, the nearby inflammatory process probably leads to early thrombosis of the pseudoaneurysm in a majority of cholecystitis-related cases, which may explain the rarity of this entity (4,lO). The etiology of the pseudoaneurysm in this patient is unclear. There was no evidence of gallstones or acute cholecystitis. Pancreatitis, possibly drug-related, may have been the causative factor as the only nearby inflammatory process, but also could have occurred as a result of hemobilia, with an obstructive clot at the papilla of Vater. Eighty percent of patients with pseudoaneurysms of the hepatic artery or its branches present with bleeding, which ranges from microcytic anemia or melena to cata-
strophic hematemesis. Rupture occurs in the biliary tract in 45% of patients, usually in the gallbladder, and on to the common bile duct. Hemobilia leads to the complete Quincke's triad (biliary colic, obstructive jaundice, gastrointestinal bleeding) in only 32% of patients, making it a difficult clinical diagnosis (2,12,15). Most patients presenting with hemobilia are likely to undergo emergency gastroduodenal endoscopy and, therefore, will incur the risk of iatrogenic sclerosis of the ampulla, mistaken for a bleeding ulcer, as occurred for this patient. Useful diagnostic tools for pseudoaneurysms of the cystic artery or other branches of the hepatic artery, include spiral CT and digital substraction celiac arteriography, which will reveal the aneurysmal dilation and the contrast material extravasation. US is likely to be performed in these patients, but may at most suggest the diagnosis if a high index of suspicion is present. The diagnostic value of arteriography is enhanced by the possibility of performing a therapeutic procedure. In opposition to pancreatitisrelated pseudoaneurysms, which are most frequently located in the pancreaticoduodenal arcades or the splenic artery, and where embolization should only be a temporary option before surgical management (16,171, embolization of a postcholecystectomy pseudoaneurysm located
in the stump of the cystic artery can probably be considered as a definitive therapeutic modality, if successful. However, one should be aware that failures of embolization or selective catheterization of the feeding vessel may occur and may be fatal (4,14). In cases of cholecystitis-related pseudoaneurysm, urgent cholecystectomy is obviously the treatment of choice. Nevertheless, if the diagnosis is revealed by arteriography, transcatheter embolization should be attempted because it only slightly prolongs the duration of the procedure, permits the delay of intervention in unstable patients with shock or sepsis, allows bleeding control during operation, and does not impede surgical procedures (1,12,14).During surgery, identification and ligation of the aneurysmal cystic artery in an inflammatory territory may be difficult, especially if bleeding is serious and uncontrolled. In such cases, ligation or temporary clamping of the right hepatic artery may be life-saving (4). In summary, pseudoaneurysms of the cystic artery are rare and a high index of suspicion is required for identification. Digital substraction arteriography is the best diagnostic option and allows transcatheter embolization, either as a definitive or temporary therapeutic procedure. Surgery is required when the origin is an inflammatory process, such as cholecystitis, or in the case of failure of embolization.
792
Embolization of Cystic Artery Pseudoaneurysm
June 1999 JVIR
References 1. Baker KS, Tisnado J, Cho SR, Beachley MC. Splanchnic artery aneurysms and pseudoaneurysms: transcatheter embolization. Radiology 1987; 163:135-139. 2. Maw A, Mander BJ, Nandi SC, Taube M, Evans HJR. Pseudoaneurysm of the cystic artery. Eur J Surg 1997; 163:307-309. 3. Bergey E, Einstein DM, Herts BR. Cystic artery pseudoaneurysm as a complication of laparoscopic cholecystectomy. Abdom Imaging 1995; 20:75-77. 4. Barba CA, Bret PM, Hinchey EJ. Pseudoaneurysm of the cystic artery: a rare cause of hemobilia. Can J Surg 1994; 37:64-66. 5. Ledermann HP, Schoch E, Jost R, Decurtins M, Zollikofer CL. Superselective coil embolization in acute gastrointestinal hemorrhage: personal experience in 10 patients and review of the literature. JVIR 1998; 9:753-760. 6. Devin R, Mambrini A, Winniger A, et al. Un cas d'ankvrysme de
7.
8.
9. 10.
11.
12.
l'arthre cystique rompu dans la vksicule biliaire. Chirurgie 1971; 97:332-335. Hakami M, Beheshti GH, Arnirkhan A. Hemobilia caused by a rupture of cystic artery aneurysm. Am J Proctol 1976; 27:56-57. Reddy SC. Pseudoaneurysm of cystic artery with upper gastrointestinal hemorrhage. South Med J 1983; 76:85-86. Rhee JW, Bonnheim DC, Upson J . Cystic artery pseudoaneurysm. NY State J Med 1987; 87:47. Wu TC, Liu TJ, Ho YJ. Pseudoaneurysm of the cystic artery with upper gastrointestinal hemorrhage. Acta Chir Scand 1988; 154:151-152. Smague EA, Schulte F, Guse S. Recurrent hemobilia caused by a ruptured pseudoaneurysm of the cystic artery in the gallbladder. Chirurg 1990; 61:199-200. Clements WDB, Wilson RH, Crothers JG, McIlrath EM, Johnston GW. Pseudoaneurysm of the cystic artery following cholecystectomy. J R Coll Surg Edinb 1993; 38:348-353.
13. Zilberstein B, Cecconello I, Cardoso Ramos A, Sallet JA, Andersen Pinheiro E. Hemobilia as a complication of laparoscopic cholecystectomy. Surg Laparosc Endosc 1994; 4:301303. 14. Stewart BT, Abraham RJ, Thomson KR, Collier NA. Post-cholecystectomy heemobilia: enjoying a renaissance in the laparoscopic era? Aust N Z J Surg 1995; 65:185-188. 15. Winchester DP, Seed RW, Bergan J J , Conn J J r . Jaundice, hemobilia, and hemoperitoneum: consequences of rupture of hepatic artery aneurysm. Am J Surg 1970; 120: 384-387. 16. Lee MJ, Saini S, Geller SC, Warshaw AL, Mueller PR. Pancreatitis with pseudoaneurysm formation: a pitfall for the interventional radiologist. AJR 1991; 156:97-98. 17. de Perrot M, Berney T, Delgadillo X, Buehler L, Mentha G, Morel P. Management of bleeding pseudoaneurysms in patients with pancreatitis. Br J Surg 1999; 86:2932.