Introduction
Aneurysmal disease of visceral arteries is uncommon, and it is usually involved with branches of the hepatic and gastro-duodenal arteries.[1] We report a patient with upper abdominal pain,malaena and jaundice due to hemobilia, caused by cystic artery aneurysm in the context of blunt abdominal trauma.
Case Report.
A 25-year-old male patient was sent to the Emergency Department of this hospital, with a sudden onset of acute abdominal pain originating from the right upper quadrant (RUQ). Loss of appetite, nausea, vomiting, hematemesis were not observed. The patient had a history of blunt trauma abdomen following a Road Traffic Accident 6 weeks previously following which he was admitted in a local hospital where he received treatment in the intensive care unit for two days becuse he was unconscious and was hemodynamically unstable.Emergency CT Abdomen revealed:Grade 3-4 liver injury;B/L lower lung hemmorhage and right adrenal hemmorhage. There were no features suggestive of cholecystitis. [Figure 1]
Figure 1: CECT Abdomen at time of trauma : shows Grade ¾ Liver laceration
He developed malaena and jaundice during 1st week of his hospital stay. The patient was treated conservatively and his malaena and jaundice subsequently subsided. Serial USG while in hospital showed resolving contusion of liver.He was discharged after three weeks of hospital stay when his hemodynamic status was stable and blood parameters were within normal limit.
The patient presented with episode of severe abdominal pain and malaena one week after his discharge and hence was readmitted in the same hospital.Repeat CECT abdomen showed:Resolving contusion in left lobe of liver; mild-moderate IHBR [Figure 2]; Dilated CBD due to obstruction by non-enhancing dense mass in CBD-Blood clots [Figure 3]; Intra luminal blood clots in GB.
Figure 2: CECT Abdomen at time of jaundice and hemobilia : shows dilated common bile duct and intrahepatic biliary radical
Figure 3 : Pre Procedure Angiogram: shows pseudoanurysm of cystic artery
He also had a low Hb value of 4 for which he was transfused with 2 points of whole blood.
The patient was then referred to our hospital.His Hb was 8.9 on admission and his S.Bil(T) was 2.8 and direct component 1.2.
He was still complaining on malaena and was having intermittent episodes of severe abdominal pain,predominantly right upper part. Subsequent blood investigations revealed Hb falling from 8.3 to 6.9 following which the patient was transfused with 1 point of whole blood.His Hb value then stabilised at 8.9 for two consecutive values.S.Bil also showed a rising trend with values reaching 3.5(T) and 2.5 as the direct component.S.Alkaline phosphatise also increased from admission value of 554 to 1646 subsequently.
USG abdomen taken in our Radiology Dept revealed Resolving contusion left lobe of liver;GB distended with echogenic material suggestive of blood clots and intraluminal blood clots in GB.Patient was then subjected to CT Angiogram of abdomen in our Radiology dept which showed a cystic artery pseudoaneurysm with features of chronic cholecystitis and resolving contusion of liver.
DSA and embolization was planned for this patient which showed pseudoaneurysm in the cystic artery branch [Figure 4]. Embolization was done successfully for the patient using two Bernstein liquid coils.
Figure 4 : Post Procedure Angiogram : shows endocoil insitu
Post procedure DSA showed complete occlusion of the aneurysm with no flow distally [Figure 5].
The condition of the patient remained clinically stable and follow-up showed normal hemoglobin and S.Bilirubin levels. The patient was discharged after 7 days with a plan for outpatient follow-up.
DISCUSSION
Cystic artery pseudoaneurysm is a rare disease, such that an extensive search identified only 27 published cases. Pseudoaneurysms are differentiated from true aneurysms by the presence of a known cause such as trauma or inflammation, and by radiologic findings of aneurysmal dilatation in the setting of otherwise normal artery. Causes of cystic artery pseudoaneurysms include abdominal trauma and intraabdominal inflammatory processes such as cholecystitis or pancreatitis.[1] CAP is invariably associated with inflammation of the gallbladder[13,14,15].The etiology of aneurysm in our patient is blunt abdominal trauma.
The above table shows cases of cystic artery aneurysms secondary to inflammatory causes and our case represents the first case of cystic artery pseudoaneurysm causing hemobilia post trauma.
Cystic artery pseudoaneurysm usually manifests itself with hemobilia, hematemesis and/or melena.[3,4,5] First described in 1654 by Francis Glisson, hemobilia commonly presents as an upper gastrointestinal hemorrhage.[6,7] Causes of hemobilia can be traumatic and non-traumatic. The most common traumatic cause in Western countries is liver trauma, followed by iatrogenic injury during diagnostic and therapeutic hepatobiliary procedures.[6] Non-traumatic causes include cholecystitis and pancreatitis.[8,9] Hemobilia is classically associated with Quincke's clinical triad of colicky abdominal pain, jaundice and gastrointestinal hemorrhage.[5] Because of the rarity of this disease, there is no consensus on the clinical management or operative treatment of cystic artery pseudoaneurysms. While cholecystectomy is considered as the definitive treatment in most cases, arterial embolization has also been used effectively to treat visceral artery aneurysms including cystic artery aneurysms.[10]
Some have combined cholecystectomy with arterial embolization.[11] Cholecystectomy may specifically be considered if (necrotizing) cholecystitis is suspected. In the past there was a concern that embolization of the cystic artery would cause gallbladder necrosis, but recent publications invalidate this theory.[9]
Arterial embolization has the advantages of reduced surgical morbidity and mortality compared to open or laparoscopic cholecystectomy. The port for catheter insertion at the femoral artery can be left in place for 24 hours in case there is rebleeding and there is a need for re-evaluation using an angiogram. In addition, the length of hospital stay can be shorter, reducing the cost of treatment. The treatment of all the reported cases of cystic artery aneurysm is shown in Table. While promising, the angiographic treatment is not possible in all cases. Many of the reported cases had associated cholecystitis and were surgically managed with cholecystectomy with operative repair of aneurysm. Morioka et al[12] reported a patient in whom arterial embolization could not be performed since the diameter of the aneurysm was too small to embolize, and therefore cholecystectomy and aneurysm repair was performed.Some cases were treated with embolization combined either with post cholecystectomy or aneurysm repair. One paper reported a patient with idiopathic aneurysm treated with embolization only.
Conclusion
Visceral artery aneurysms are rare and can rupture with potentially grave outcome due to excessive bleeding. Cross-sectional imaging usually demonstrates the pathological changes of the disease. If a cystic artery pseudoaneurysm is demonstrated, microcoil embolization appears to be the most appropriate management.[16]
Angiographic embolization as a common method of treatment for visceral artery aneurysms was successfully used in our patient. This represents the first documented case of post traumatic cystic artery pseudoaneurysm leading to hemobilia.
References
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9. Hepatobiliary Pancreat Dis Int,Vol 7,No 5 • October 15,2008
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