Abbreviations:

VAA Visceral artery aneurysm; HAA Hepatic artery aneurysm; CT Computed Tomography; ERCP Endoscopic Retrograde Cholangiopancreatography; EUS Endoscopic Ultrasound

1. Introduction

The hepatic artery aneurysm (HAA) is a rare cause of cholestasis. There are also publications describing mechanical jaundice caused by the compression of aneurysms originating from other arteries. It is the right and left branch of the hepatic artery, gastroduodenal artery, pancreatoduodenal arteries, celiac trunk, and arc of Buhler as in Table 1. The course of HAA, even in 75% of cases is asymptomatic. 14% of cases are complicated by rupture and hemorrhage [1]. Before the development of imaging techniques such as computed tomography or ultrasound, the diagnosis of VAA was stated on the ground of the autopsy or during the laparotomy [2-4]. Today a detection of an aneurysm is more often earlier, which enables successful treatment [5,6]. 92.4% of VAA are diagnosed accidentally during imaging in asymptomatic patients [7]. The first case of HAA that caused mechanical jaundice was described by Stokes in 1834 [2]. The diagnosis was made based on an autopsy. The first successful treatment was carried out by Kher in 1903 by ligating the HAA [4]. It is assumed that the first preoperative diagnosis through angiography was described by Doppman in 1963 [8]. In contrast, the first description of the embolization of a visceral aneurysm causing mechanical jaundice was published in 1994 [9], although an embolization of aneurysms has been performed since the 1970s. Endovascular, surgical, and alternative methods are used to treat VAA. In endovascular methods, thrombin, vascular adhesives, onyx, or spirals are administered to the aneurysm causing the aneurysm to clot or the stents can be inserted into the artery to cut off the blood supply to the aneurysm and preserve blood flow. Surgical methods are based on: ligation of the vessel with an aneurysm, aneurysmorrhaphy, excision of an aneurysm with anastomosis of the ends of the artery directly or with the use of a prosthesis as well as vein graft. Sometimes it is necessary to ligate vessels with an aneurysm with the formation of a bypass or to remove an aneurysm with a fragment of an organ. [6,10-13] Alternative methods to the above-mentioned are: percutaneous embolization of the aneurysm under ultrasound or CT control and embolization with the usage of EUS [13,14]. Due to the rarity of VAAs, reports on them are limited to case studies and case series [11]. This makes it difficult to determine the best therapeutic path for the choice of technique and timing of intervention [10]. There is no consensus on the optimal treatment of VAA [12]. Endovascular management is the treatment of choice, however, often due to unfavorable morphological conditions of the aneurysm, surgery remains the best or only solution [14]. According to the European Society of Vascular Surgery guidelines from 2017, all symptomatic VAA require repair regardless of their size and location. Asymptomatic patients with VAA <25 mm may be followed up with imaging control every 2-3 years. Invasive treatment should be considered for asymptomatic aneurysms> = 25 mm [15]. The endovascular procedure should be planned first. If it is technically possible and the patient does not present a high perioperative risk, the procedure should be aimed at the reconstruction of the vessel and restoration of blood flow through the vessel. Patients in poor general condition and at high perioperative risk with aneurysms between 2 cm and 5 cm may be observed, but each case should be assessed on an individual basis. Aneurysms larger than 5 cm should be surgically treated [1]. Regardless of the size of the aneurysm, the intervention should be offered to women of childbearing age, planned liver transplant recipients, patients with pancreaticoduodenal, gastroduodenal, or intrahepatic aneurysms. In the case of unstable or uncontrolled hypertension, an aneurysm enlargement, or an arteriovenous fistula, the patient should be qualified for the intervention [1,15]. Visceral Artery Pseudoaneurysms, due to the high risk of perforation, should be provided urgently [14,15]. As reported by Pitton and ca. in a 10-year retrospective single-center analysis on 233 patients - no significant difference in diameter was observed between ruptured and unruptured visceral aneurysms, therefore the author suggests that the qualification should not be based solely on the size [7].

2. Case Presentation

A 93-year-old woman with painless jaundice was admitted to the General Surgery Department. Except for the compensated arterial hypertension, the patient suffered no other significant comorbidities.

The physical examination of the bedridden elderly patient stated no deviation except jaundice.

The laboratory blood tests revealed: elevated total bilirubin level 258,4 umol/L (n. 5.0-21.0 umol/L), higher activity of aminotransferases AST - 318 U/L (n. 0-35 U/L), ALT - 555 U/L (n. 0-35 U/L) and GGTP - 370 U/L (n. 0-38 U/L) with INR 1,20 (n. 0.80 - 1.20). On the ground of ultrasonographic doppler of the abdomen, cholestasis caused by hepatic artery aneurysm was stated.

Figure 1: Ultrasonographic finding in the hilum of the liver.

The diagnosis was confirmed by CT angiography. The fusiform aneurysm 85 mm x 40 mm was located under the liver and in the hilum. The aneurysm compressing gallbladder and common bile duct consisted of proper hepatic artery aneurysm (24.2 mm diameter and 38.6 mm length) and right hepatic artery aneurysm (36.3 mm diameter and 51.3 mm length). The celiac trunk was absent, splenic artery and left gastric artery arose directly from the abdominal aorta. The common hepatic artery branched from the superior mesenteric artery. Enlargement of the abdominal aorta to 34 mm with an intraluminal thrombus 7 mm thick on a length of 50 mm was also described.

Figure 2: Proper hepatic artery aneurysm and right hepatic artery aneurysm in CT angiography of the splanchnic vessels.

To restore bile flow ERCP with papillotomy and stent (8.5 F 10 cm) placement inside the common bile duct was performed. The procedure was successful, and reduction of the total bilirubin level to 60.3 umol/l [n. 5.0 - 21.0] was noticed.

Figure 3: Stent placement during ERCP.

The postoperative course was complicated with upper gastrointestinal bleeding. An Esophagogastroduodenoscopy was performed and a source of the bleeding was found on the posterior wall of the stomach. The hemobilia was excluded and the hemorrhage was stopped with hemostatic clips. There were no other symptoms of gastrointestinal bleeding during hospitalization. Because of the aneurysm's size and anatomy, the patient was disqualified from the endovascular procedure by two vascular surgeons. Open surgery was proposed but the patient didn’t agree to the suggested treatment. She was discharged home with recommended ambulant control and USG of the abdomen in 2 months. The patient died at home a couple of weeks after discharge

3. Discussion

Along with the aging of the population and associated increasing numbers of patients aged over 90, surgeons face the problem of acute intervention in geriatric patients. The data show high perioperative mortality in this age group of 11% in the case of emergency surgery [16]. In line with the above, the treatment of the patient we described was planned in two stages. In the first stage, endoscopic biliary drainage was done. In the second stage, treatment of the aneurysm was planned. After biliary drainage was successful, the vascular procedure could be performed electively. Morphology of aneurysms - size, and vascular anomaly - arising of the hepatic artery from the superior mesenteric artery, high risk of aneurysm dissection, and gastrointestinal bleeding during the procedure were the reasons for the patient's disqualification from endovascular treatment. The patient did not consent to the open surgery, which could cure the underlying disease, however, taking into account the cardiovascular capacity and sickness burden, it could probably accelerate the death. The patient survived hospitalization but died a couple of weeks after discharge. The autopsy was abandoned, therefore the cause of death cannot be determined. Most authors do not follow up, therefore the long-term effects of therapy or the fate of patients after hospitalization are often impossible to determine. If the time of discharge is taken as the cut-off point, as in most publications, the patient survived. Three similar cases have been reported in the literature. Due to the lack of consent to the vascular procedure, the intervention in the patient described by Julianow was also limited to ERCP with a biliary stent. After 12 months, a follow-up was performed, the patient did not present any complaints. The originally described HAA was 64 mm in diameter [17]. The patient presented by Rathiel was disqualified from the vascular procedure due to cardiovascular insufficiency, while endovascular embolization was abandoned due to concerns about extensive ischemia of the treated area [18]. There is no information regarding the patient's fate after the discharge. The patient presented by Peter did not make it to the procedure scheduled for the next day. Death was due to massive hemorrhage [19]. The literature review was performed using the database "Pub Med" using the keywords "jaundice" and "aneurysm,". To identify additional potentially relevant data sources, we hand-searched the reference lists of the retrieved studies. Reports from 1834 to 2021 concerning mechanical jaundice caused by an aneurysm of visceral arteries were selected. The analysis included studies in which it was possible to determine the procedure, treatment results, and the artery with which the aneurysm was involved. In the reviewed works from 1834 to 2021, 77 cases were described. The oldest case so far was an 89-year-old woman, while the oldest men with this condition were 85 years old. The literature review is summarized in Table 1 and Table 2. The conservative treatment or surgery without vascular procedure in the case of VAA complicated by jaundice almost always resulted in the patient's death. In 28 cases only 1 survival was recorded. The only case with a patient's survival was described by McEwan-Alvarado. There is no information about follow-up [8]. ERCP without other procedures was performed in 4 patients, 3 of them were discharged, and one died during hospitalization. The endovascular treatment was used in 28 cases, in 10 cases it was preceded by ERCP. There was no mortality after endovascular treatment. Ten patients required vascular surgery after an intravascular treatment. In the case of the surgical vascular procedure 5 patients out of 28 died.

Table 1: Literature review

Abbreviations: HA Hepatic artery; RHA right hepatic artery; LHA left hepatic artery; GDA gastroduodenal artery; PCDA pancreatoduodenal artery; CA celiac axis; SMA superior mesenteric artery; PSA pseudoaneurysm; ERCP Endoscopic Retrograde Cholangiopancreatography; PTC Percutaneous transhepatic cholangiography; TAE transcatheter arterial embolization.

Table 2: Treatment procedure

4. Conclusion

Jaundice due to HAA can be observed in patients over 90 years of age, so this possibility should be taken into account in the differential diagnosis. VAA with jaundice requires an endovascular or surgical intervention. In hemodynamically stable cases ERCP with biliary stenting can be performed before causative endovascular or surgical treatment, to prepare the patient for further treatment. ERCP with biliary stenting can also be performed in the absence of the patient's consent to the vascular procedure to protect the liver function.

Funding:

None

Conflict of interest:

None declared

Ethical Approval:

Not required

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