Hepatic artery or cystic artery pseudoaneurysms are rare complications of laparoscopic cholecystectomy, with cystic artery involvement being reported much less frequently in the literature. It is hard to determine the incidence of the pseudoaneurysm, as it could be asymptomatic, thrombosed or ruptured [2, 3]. The reported time interval between the surgery and onset of clinical symptoms is variable, with one month reported as the average time to presentation. However, a 5-year delay in presentation has been described [2].
The exact mechanisms of hepatic or cystic artery pseudoaneurysm after laparoscopic or open cholecystectomy is not completely identified up to date, however, they can be related to direct vascular injury (by applying surgical clips/ thermal injury), adjacent gallbladder fossa bile collection that increases vessel wall fragility or as a consequence of post-surgical adhesions. Most of the cases presented with classical symptoms of haemobilia (gastrointestinal bleeding, upper abdominal pain, and jaundice) [4], but it is rare to present with symptoms of lower GI bleeding [5].
Pseudoaneurysm is considered an acute emergency that requires immediate intervention. The diagnosis can be made by endoscopy, ERCP, CT and conventional angiography, and managed primarily by TAE through occluding the sac or the feeding vessel with a variety of embolic agents, including coils, thrombin, or gel foam, before ideally embolizing the vessel distal and proximal to the pseudoaneurysm to prevent its collateral filling [4]. Yet, surgical resection of the pseudoaneurysm and ligation of the cystic artery (CA) stump or right hepatic artery (RHA) would be an alternate treatment option if TAE fails.
Up to the authors’ knowledge, in the last five years, 46 cases of porta hepatis pseudoaneurysm were reported [2, 4, 6–24] (Table 1). 52.1% (24 cases) were males, age range from 39 – 82 years, with mean age of 56.3 years, and 47.9% (21 cases) were females, age range from 37- 88 years and mean age of 61.1 years. The most common involved vessel was RHA (37 cases, 80.4%), followed by CA (8 cases, 17.4%), while left hepatic artery involvement was reported in one case. 41 cases were managed through laparoscopic approach while 5 cases were done through open resection and time of presentation ranged from day 1 up to 26 months post-procedure (mean of 60.4 days). The most common documented presentation was haemobilia, as reported in 31 cases (78.3%). Furthermore, abdominal pain, sepsis, jaundice, and hypotension were encountered, with only one case in the last five years presented with hematochezia. In 12 cases, iatrogenic bile duct injury was considered, however, no case documented the presence of bile collection/biloma. 2 out of 46 cases (4.3%) experienced an increased pseudoaneurysm size, however, 4 cases confirmed recurrence of bleeding episode (8.7%). 41 cases (89.1%) were successfully managed by TAE, while 3 cases (6.5%) required surgical ligation, 2 cases (4.3%) treated by percutaneous direct puncture and one case (0.1%) managed conservatively. Favorable outcome was achieved in 97.8% of reported cases.
In this case, we are discussing an unusual presentation and recurrence of CASP bleed, in which, the three assumed pathological factors for cystic artery/ RHA pseudoaneurysm formation are present, including vascular injury by surgical clips, post LC bile collection and extensive post-surgical adhesions. In the related literature review, only one similar presentation of fresh bleeding PR secondary to CASP was identifie. Furthermore, the source of hematochezia in our case was difficult to localize by different investigative modalities. Accordingly, surgical intervention was the definite treatment.
Table 1
Literature review of pseudoaneurysms of cystic artery or hepatic arteries following cholecystectomy
Author/ Year of the article | Number of cases | Age/ Gender | Type of surgery | Time to presentation | presenting complaint | Bile duct Iatrogenic injury | Vessel involved | Increased aneurysms' size | TX | Recurrence | adhesions |
Rossini M et al., 2019 [2] | 1 | 66/M | OC | 28 Days | RUQ pain, Hemobilia | No | CA | No | TAE/Coils | No | No |
Machado et al, 2017 [4] | 1 | 70/F | LC | 14 Days | Abdominal pain | No | RHA | No | TAE/Coils | No | No |
Gachabayov M et al., 2017 [12] | 1 | 57/M | LC | 15 Days | Hemobilia, jaundice, abdominal pain | No | RHA | Yes | TAE/Alcohol particles + Surgical ligation | Yes | No |
Wen F et al., 2016 [23] | 14 | 49 m/ 10 F, 4M | LC | 21 Days (m) | Hemobilia, abdominal pain, jaundice | No | RHA | No | TAE/Coils | No | No |
CreTu O M et al., 2017 [11] | 1 | 55 | LC | 22 Months | Haemobilia + upper abdominal pain | No | RHA | No | Surgical ligation | No | No |
Ion D et al., 2016 [14] | 1 | 58/M | OC | 27 Days | Haemobilia+ anemia + hematochezia | No | RHA | No | TAE | Yes | No |
To K et al, 2018 [20] | 1 | 56/M | LC | 28 Days | Haemobilia +RUQ pain | No | CA | No | TAE/Coils + stent | No | No |
Abiko T et al., 2020 [6] | 1 | 60/M | LC | 3 Days | Haemobilia + RUQ pain | No | CA | No | TAE | No | No |
Choudhary A et al., 2017 [10] | 1 | 42/F | LC | 26 Months | RUQ pain+ hematemesis | No | CA | No | TAE/coil + stent/distal bulge | No | No |
Villa-Gomez G et al., 2018 [22] | 1 | 39/M | LC | 35 Days | Hematemesis+ cholangitis | Yes | RHA | No | TAE | No | No |
Badillo R,et al., 2017 [9] | 1 | 79/M | LC | 15 Months | Haemobilia | No | CA | No | PDP | No | Yes |
Arata R et al., 2020 [8] | 1 | 88/F | LC | 12 Days | Haemobilia +Abdominal pain+ Back pain | No | CA | No | conservative | No | No |
Traa AC et al., 2020 [21] | 1 | 76/F | LC | 1 Day | Pain+ haemobilia | No | RHA | No | TAE/Coil+ stent/coil | Yes | Yes |
Tiwari A et al., 2017 [19] | 1 | 80/M | LC | 30 Days | UGIB+ hematochezia+ shock | No | LHA | No | TAE | No | No |
Yagihashi K et al., 2017 [24] | 1 | 55/M | OC | 7 Days | Fever + abdominal pain | No | RHA | No | PDP | No | No |
Rege S A et al., 2017 [17] | 1 | 40/F | LC | 55 Days | Hematemesis + RUQ pain | No | CA | No | TAE/Coils | No | No |
Alrajraji M et al., 2016 [7] | 1 | 41/F | LC | 8 Months | Hematemesis + melena (haemobilia) | No | RHA | Yes | Surgical ligation | Yes | No |
Gandhi R J et al., 2020 [13] | 13 | 52m. 3F, 10M | LC | 9-30 Days (15.6days=m) | 3 hematemesis, 2 Malena, 4 sepsis + anemia. 4 hypotension | Yes (9 cases) | RHA | N/A | TAE(3Coils,4NBCA,6Coil+NBCA) | No | No |
Kassem T W et al., 2017 [15] | 1 | 42F | LC | 6 Months | Hematemesis | No | RHA | No | TAE/Coils | No | No |
Rosa C et al., 2018 [18] | 1 | 52F | OC | 4 Months | Hematemesis + Malena (haemobilia) | Yes | RHA | No | TAE/NBCA | No | Yes |
Mahfooz F et al., 2020 [16] | 1 | 82M | OC | 2 Months | Abdominal pain + Malena | Yes | CA | No | TAE | No | No |
CA: Cystic artery, RHA: Right hepatic artery, LHA: Left hepatic artery, TAE: Trans-arterial embolization, NBCA: n-butyl-2-cyanoacrylate, PDP: percutaneous direct puncture, LC: laparoscopic cholecystectomy, OC: open cholecystectomy, M: Male, F: Female, m: Mean, RUQ: right upper quadrant pain
CASP is a rare complication of post LC, yet a potentially life threating, with possible delayed complications occurring months to years after the surgery. We are presenting a rare case of CASP presented with melena and fresh bleeding per rectum, with presence of all possible etiopathogenesis, diagnosed by contrast CT and angiogram, and eventually treated with surgical ligation after failure of the initial TAE. Clinicians and radiologists should be aware of this important entity and its variable manifestations to facilitate early treatment.