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| Introduction The mainstay of treatment of cystic artery pseudoaneurysm is cholecystectomy and ligation. Recent reports have shown success in using microcoil embolization.[1-5] We highlighted an unusual mode of presentation in a case, and used percutaneous thrombin injection and coil embolization as definitive treatment methods. Case report A 63-year-old man was admitted to a district general hospital following an acute onset of epigastric pain and hematemesis. He had an 18-year history of rheumatoid arthritis and was on methotrexate and nonsteroidal antiiflammatory drugs. Upper gastrointestinal endoscopy showed severe reflux oesophagitis and he was commenced on proton pump inhibitor. Four days later the patient developed acute peritonism. At laparotomy 1700 ml blood was found in the peritoneal cavity. The gallbladder was noted to be firm and tense, but despite a thorough laparotomy no obvious source of bleeding was identified. Abdominal drains were sited and the patient's general condition improved postoperatively. Twelve days later the patient developed recurrence of abdominal pain and peritonism. At the second laparotomy the gallbladder was tensely distended and there was again more than a litre of blood in the peritoneal cavity. A cholecystectomy was performed. The gallbladder was found to contain blood clots but no stones. The subsequent histopathological report revealed evidence of acute or chronic cholecystitis. In addition, an erosive cavity measuring 2×3 cm in size was detected at the porta hepatis. There was no active bleeding and there was 1400 ml of blood stained fluid in his drain over the following 48 hours. Four units of blood were required to maintain his level of hemoglobin above 8 g/dl. Three days later the patient became jaundiced with elevation of bilirubin and alkaline phosphatase levels. Ultrasound examination of the abdomen suggested an extrahepatic bile duct obstruction. Endoscopic retrograde cholangiopancrea-tography (ERCP) showed no stones. An abdominal CT scan demonstrated a central intrahepatic lesion at the site of the gallbladder fossa. At this stage the patient was referred to our tertiary hepatobiliary center. On admission, he was pyrexial and jaundiced with tenderness in the right upper quadrant. The abdominal drainage was still blood stained. Angiography was performed for suspected hemobilia and clearly demonstrated pseudoaneurysm of the cystic artery as the source of hemoperitoneum (Fig.1). The pseudoaneurysm was injected percutaneously with 500 IU of thrombin. Successful thrombosis of the sac was confirmed with colour Doppler immediately after the procedure and again four days afterwards. At discharge, the abdominal drainage had ceased completely and the patient was afebrile and hemodynamically stable. A week later the patient was readmitted with melaena and hypovolaemic shock. After resuscitation, an emergency angiogram demonstrated recurrent hemorrhage from the cystic artery pseudoaneurysm. Metal microcoil embolization of the pseudoaneurysm was performed and the bleeding stopped (Fig.2). The patient made a slow but uneventful recovery over the following 10 days and was discharged. Follow-up Doppler study at 6 weeks showed resolution of the aneurysm sac. The patient remains asymptomatic at three years and has now been discharged for further follow-up.  Discussion Spontaneous hemoperitoneum of hepatobiliary origin is most often caused by bleeding from liver tumors, and in our unit we deal with this most usually by embolization as a primary maneuver. Rupture of aneurysms arising from the left gastroepiploic artery, the hepatic artery, the middle colic artery and the superior mesenteric artery has been reported.[2-5] Cystic artery pseudoaneurysm (CAP) is a rare clinical entity. Our literature search revealed fewer than 20 cases had been reported to date. CAP is invariably associated with inflammation of the gallbladder.[6-8] Most of the reported cases presented with hemobilia alone.[7,8] The patient in the current report presented mainly with hemoperitoneum and some evidence of hemobilia. We could find only one other similar report in the literature. In the former report, aneurysmal dilatation of the cystic artery was present in addition to multiple aneurysms of the left and right hepatic arteries. The exact source of the hemoperitoneum however was not conclusively proven.[6] This paper therefore represents the first definitive report of hemoperitoneum being caused by CAP. The rarity of the lesion probably accounts for the delay in diagnosis. Hemorrhage from the cystic artery usually presents as hemobilia and is usually attributed to erosion by gallstones, particularly in the elderly. Inflammation of the gallbladder may lead to similar changes in the cystic artery and may rarely lead to aneurysm formation. The aneurysm may then erode into the common bile duct or gallbladder, and rarely as in our case into the peritoneum. This accounts for the presentation with hemobilia and bleeding into the peritoneal cavity. Color Doppler has been used successfully to diagnose CAP.[6] However, in our patient an angiogram showed the acute presentation. Surgical ligation of the aneurysm sac with cholecystectomy has been the most commonly used treatment modality. A recent report[9] has also demonstrated successful treatment of CAPs with metal coil embolization. In our patient, percutaneous thrombin injection into the aneurysm sac was done initially. This maneuver was based on the experience of the radiologists who had used this technique successfully in obliterating a pseudoaneurysm of the hepatic artery in a patient after liver transplantation. However, in our patient, percutaneous thrombin failed after initial success. The aneurysm was finally obliterated successfully using microcoil embolization. In conclusion, our experience with this case suggests that if no cause can be found at emergency laparotomy for hemoperitoneum, cholecystectomy should be carried out and angiography should also be performed to exclude a visceral artery aneurysm that may have been missed. If a cystic artery pseudoaneurysm is demonstrated, microcoil embolization appears to be the most appropriate management. Funding: None. Ethical approval: Not needed. Contributors: GA, KE and KA wrote the first draft. HK summarized patient’s history. KD, PJ and LJPA edited the manuscript. All anthors contributed to the intellectual context and approved the final version. GA is the guarantor. Competing interest: No benefits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article. References 1 Ong GB, Taw JL. Spontaneous rupture of hepatocellular carcinoma. Br Med J 1972;4:146-149. PMID: 4342761 2 Jacobs PP, Croiset van Ughelen FA, Bruynickx CM, Hoefsloot F. Haemoperitoneum caused by dissecting aneurysm of the gastroepiploic artery. European J Vascular Surg 1994;8:236-237. PMID: 8181623 3 Noah EM, Psathakis D, Bruch HP, Kagel C. Perforated aneurysm of the left hepatic artery. Zentralbl Chir 1992;117:556-560. PMID: 1441780 4 Borioni R, Garofal M, Innocenti P, Fittipaldi D, Tempesta P, Col Seddio F, Pace A. Haemoperitoneum due to spontaneous rupture of an aneurysm of the left gastroepiploic artery. J Cardiovasc Surg 1999;40:63-64. PMID: 10221388 5 Blumenberg RM, David D, Slovak J. Abdominal apoplexy due to rupture of a superior mesenteric artery aneurysm: clip aneurysmorrhaphy with survival. Arch Surg 1974;108:223-226. PMID: 4810534 6 Kaman L, Kumar S, Behera A, Katariya RN. Pseudoaneurysm of the cystic artery: a rare cause of haemobilia. Am J Gastroenterol 1998;93:1535-1537. PMID: 9732939 7 England RE, Marsh PJ, Ashleigh R, Martin DF. Case report: pseudoaneurysm of the cystic artery: a rare cause of haemobilia. Clinical Radiol 1998;53:72-75. PMID: 9464443 8 Barba CA, Bret PM, Hinchey EJ. Pseudoaneurysm of the cystic artery: a rare cause of haemobilia. Canadian J Surg 1994;37:64-66. PMID: 8306223 9 Delgadillo X, Berney T, de Perrot M, Didier D, Morel P. Successful treatment of a pseudoaneurysm of the cystic artery with microcoil embolisatiom. Vasc Interv Radiol 1999;10:789-792. PMID: 10392949 Received March 15, 2006 Accepted after revision February 12, 2007 |
Thứ Tư, 3 tháng 8, 2011
Hemoperitoneum secondary to rupture of cystic artery pseudoaneurysm
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