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Korean J Pancreas Biliary Tract. 2023;28(4):114-119

Publication date (electronic) : 2023 October 31

doi : https://doi.org/10.15279/kpba.2023.28.4.114

Bo Ram Seo¹, Hyun Tak Lee¹, Jeong Hwan Lee¹, Ah Young Lee², Sang Jong Park¹, Ju Sang Park¹, Jun-young Seo^,1

¹Department of Gastroenterology, DMC Bundang Jesaeng General Hospital, Seongnam, Korea

²Department of Gastroenterology, CHA Gangnam Medical Center, Seoul, Korea

서보람¹, 이현탁¹, 이정환¹, 이아영², 박상종¹, 박주상¹, 서준영^,1

¹분당제생병원 소화기내과

²CHA 의과학대학교 강남차병원 소화기내과

Corresponding author : Jun-young Seo Department of Gastroenterology, DMC Bundang Jesaeng General Hospital, 20 Seohyeon-ro 180beon-gil, Bundang-gu, Seongnam 13590, Korea Tel. +82-31-779-0210 Fax. +82-31-779-0929 E-mail: terryxom11@naver.com

Received 2023 June 19; Revised 2023 July 19; Accepted 2023 July 19.

Abstract

Trans Abstract

INTRODUCTION

Pseudoaneurysm is a complication that could occur in patients suffering from acute or chronic pancreatitis [1,2]. This formation, a result of pseudocyst pressure and enzymatic action, can lead to pseudoaneurysmal bleeding, which is a life-threatening condition with a mortality rate of up to 37% [3]. It has been reported that this type of bleeding occurs in 5-10% of patients who have pseudocysts associated with chronic pancreatitis [2].

The clinical manifestation of pancreatitis-associated pseudoaneurysm could vary, ranging from asymptomatic to periumbilical pain, melena, hematochezia, and hemodynamic instability. Accordingly, it is important to accurately and timely diagnose pseudoaneruysm rupture through imaging studies [4].

As conservative management of active hemorrhage due to pseudoaneurysm is known to be associated with a high mortality rate of up to 52%, interventional treatment, such as transarterial embolization (TAE), is required [2]. Although TAE is currently considered as a primary therapeutic option for patients with pseudoaneurysms associated with pancreatitis [5], there have been limited studies on the etiology of pseudoaneurysm rupture associated with pancreatitis and the outcomes of TAE. Therefore, we aimed to present a case series of patients with pseudoaneruysm rupture associated with pancreatitis, which were ultimately treated with TAE.

CASE

Here, 4 patients were diagnosed with pancreatic pseudoaneurysm and treated with TAE, whose data are summarized in Table 1 and 2.

Table 1.

Baseline characteristics and outcomes of patients diagnosed with pancreatic pseudoaneurysmal rupture treated with trans-arterial embolization

Table 2.

Characteristics of pseudonaeruysmal rupture and clinical outcomes of the TAE

1. Case 1

A 71-year-old male visited our gastroenterology department with a complaint of weight loss (10 kg over a month). He had a history of admission due to acute alcoholic pancreatitis 4 years ago in DMC Bundang Jesaeng General Hospital. He consumed alcohol daily (1 bottle of soju per day) for the past 3 years. The patient was admitted for the evaluation of weight loss. Vital signs were stable (130/80 mmHg, 70 HR/min, 36.5℃), and initial laboratory findings showed anemia (hemoglobin: 12.2 g/dL [13-17 g/dL]), elevated C-reactive protein (12.54 mg/dL [0.00-0.80 mg/dL]), and elevated amylase (61 U/L [28-100 U/L] and elevated amylase (61 U/L [28-100 U/L]) and lipase level (98 U/L [13-60 U/L]). Accordingly, abdomino-pelvic computed tomography (AP-CT) was performed, which showed chronic pancreatitis with an approximately 1.6 cm-sized pseudoaneurysm at the anterior portion of the pancreatic pseudocyst, indicating the possibility of rupture of a branch of the splenic artery (Fig. 1A). Accordingly, we decided to perform TAE as a preventive measure considering the potential risk of additional bleeding that could lead to complications such as rupture. This intervention was successfully performed without any complication (Fig. 2A). The patient was successfully discharged from the hospital with serial follow-up.

Fig. 1.

Computed tomography images of hemorrhage from pancreatic pseudoaneurysmal rupture (arrow). (A) An 1.6 cm sized pseudoaneurysm at anterior portion of pancreatic pseudocyst, located at the pancreatic body and tail portion, (B) hematoma within the pancreatic head portion, and (C) hematoma in pancreas tail.

Fig. 2.

Angiographic images of pseudoaneurysm. (A) An 1.6 cm sized pseudoaneurysm at one branch of splenic artery (arrow) (Case 1). (B) An 1 cm sized pseudoaneurysm from duodenal branch of gastroduodenal artery (arrow) (Case 2). (C) An 1.3 cm sized pseudoaneurysm in branch of left subphrenic artery (arrow) and splenic artery (Case 3). (D) A 4.2 cm sized large pseudoaneurysmal change of common hepatic artery (arrow) with active extravasation of contrast media (Case 4).

4. Case 4

A 70-year-old male patient with a past medical history of hypertension presented to the outpatient urology department complaining of left flank pain, and AP-CT was performed, which showed a prominent ampulla of Vater (AoV) (Fig. 3A) with diffuse intrahepatic duct dilatation (Fig. 3B), raising suspicion for possible AoV cancer. Endoscopic retrograde cholangiopancreatography (ERCP) was performed, and after endoscopic sphincterotomy, a villous lesion was identified at the distal common bile duct (Fig. 3C), which was confirmed as an adenocarcinoma on histologic examination. Accordingly, the patient underwent pylorus-preserving pancreaticoduodenectomy (PPPD) for the management of the cancer (Fig. 3D). However, on the twelfth day post-surgery, he began to complain of sudden abdominal distension and abdominal pain, and he started to develop a fever of 38℃. To identify the cause of the fever and abdominal discomfort, AP-CT was performed, and the patient was diagnosed with acute pancreatitis. A decision was made to maintain fasting and conservative therapy while closely monitoring his condition. However, on the fifteenth post-operative day, he suddenly had massive melena with a significant decrease in hemoglobin levels from 10.1 g/dL to 6.9 g/dL. To evaluate and manage the active bleeding, emergent angiography was performed, which showed a 4.2 cm-sized large pseudoaneurysm in the common hepatic artery, followed by therapeutic embolization (Fig. 2D). Although technical success of embolization was achieved, his systemic condition worsened on the following day, resulting in multiple organ failure, and he ultimately expired after 25 hours after the embolization.

Fig. 3.

Images of case 4 patient. (A) Computed tography shows prominent ampulla of Vater (AoV) and (B) diffuse intrahepatic dilation and common bile duct dilatation. (C) Endoscopic image of AoV mass af ter endoscopic sphincteromy, which was revealed as an adenocarcinoma in histology. (D) Gross specimen after pyloruspreserving pancreaticoduodenectomy.

DISCUSSION

Pancreatic pseudoaneurysm is a potentially life-threatening complication often associated with pancreatitis or pancreatic surgery. As the rupture of pancreatic pseudoaneurysms can lead to catastrophic bleeding and is associated with high mortality rates, clinical suspicion and early diagnosis are essential to improve patient outcomes [3].

Pseudoaneurysms are associated with pancreatitis in approximately 10% of patients and occur due to enzymatic insult to peripancreatic arteries or erosion of a pseudocyst into adjacent visceral arteries [1]. When an inflamed pancreas leaks pancreatic enzymes, it can cause the enzymatic autodigestion of vascular walls, leading to diffuse bleeding, extensive necrosis, or pseudoaneurysm formation. Additionally, a visceral artery can become embedded within the wall of a pseudocyst, transforming it into a pseudoaneurysm [2].

The splenic artery is the most commonly affected artery (60-65%) due to its contiguity with the pancreas, followed by the gastroduodenal (20-25%), pancreaticoduodenal (10-15%), hepatic (5-10%), and left gastric (2-5%) arteries [6]. In our case series, 2 of 4 patients (50%) had pseudoaneurysms affecting the splenic artery, consistent with previous findings, whereas the other 2 patients had pseudoaneurysms affecting the gastroduodenal artery or hepatic artery. Further research is needed considering that the prognosis can vary depending on the location of the arteries.

Previous studies have reported the occurrence of pseudoaneurysms arising from pancreatitis. Golzarian et al. [7] evaluated TAE as a therapeutic modality for 9 patients with 13 pseudoaneurysms secondary to pancreatitis, among whom 8 patients had chronic pancreatitis. According to domestic reports, there were 27 cases where a preceding cause could be analyzed; among them, 23 cases involved chronic pancreatitis, and 4 cases involved acute pancreatitis. Of these cases, 19 cases had a discernible cause for pancreatitis, which was chronic alcohol consumption [8]. In our case series, the etiology of 2 patients was related to acute pancreatitis (Case 1 and Case 3). The etiology of 1 patient was related to traumatic damage to the pancreas in the presence of underlying chronic pancreatitis (Case 2). On the other hand, a patient with pancreatic pseudoaneurysm rupture, which was associated with post-operation pancreatitis after pancreatic surgery (PPPD), had a poor prognosis due to a combination of several factors such as surgery, cancer, and old age (Case 4). A comprehensive review of our cases indicated that the prognosis of pseudoaneurysm rupture may be dependent on their underlying causes.

A previous study reported that conservative management of active hemorrhage due to pseudoaneurysm was associated with high mortality, and aggressive and interventional treatment is needed [2]. In the past, surgery was the only treatment for pseudoaneurysm rupture-related pancreatitis with high mortality rates ranging from 10% to 50% [9,10]. However, TAE has been used as one of the less invasive treatment options, with outcomes not inferior to those of surgery. The effectiveness of TAE has been established in handling massive bleeding from gastric and duodenal ulcers, managing visceral artery aneurysms, and dealing with postoperative hemorrhage after abdominal surgeries, with successful hemostasis in 90-92% of cases [11-13]. More recent literature recommends TAE as the preferred treatment method for patients with pancreaticoduodenal artery aneurysms. This recommendation stems from the fact that the anatomical location of these aneurysms, often deeply embedded in the pancreas tissue, complicates surgical ligation, which can result in higher mortality rates compared to TAE [14,15]. Despite the limited number of studies evaluating the efficacy of TAE in treating pancreatitis-related pseudoaneurysms, the reported success rates have ranged from 75 to 100% [3]. In our series, 4 patients were treated with TAE, and 3 of the patients were successfully treated without complications.

In conclusion, pancreatic pseudoaneruysmal rupture is a fatal and potentially life-threatening complication associated with pancreatitis and pancreatic surgery, and TAE could be used for prompt diagnosis and treatment.

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