Gastroduodenal artery aneurysm/ pseudoaneurysm: a systematic review of reported cases

Study selection and data extraction

This systematic review was conducted in accordance with a preregistered protocol (PROSPERO: CRD42023487443) and adhered to the Preferred Reporting Items for Systematic Reviews and Meta-analyses (PRISMA) reporting guidelines. A comprehensive literature search was conducted from January 2000 to October 2023 in the PubMed, EMBASE, and Google Scholar databases. Additionally, reference lists of potentially eligible studies were screened to identify further relevant articles. No language or country restrictions were imposed. Non-English manuscripts were translated using Google Translate (Mountain View, CA, USA).

The search strategy employed the keywords “gastroduodenal artery aneurysm” and “gastroduodenal artery pseudoaneurysm”. Two authors (YTL and KTC) independently assessed titles and abstracts retrieved from the search. Pertinent manuscripts were selected for full-text review. Because no randomized controlled trials or large retrospective studies on GDA were identified during the preliminary search, case series and case reports featuring patients with GDA were included. These included studies had to provide detailed information on four key aspects: clinical manifestations of GDA, diagnostic tools employed, GDA management strategies, and patient prognosis. Unpublished research, conference communications, inaccessible articles, and case reports lacking essential details were excluded from the analysis.

The following data were extracted from the included articles: first author name, publication year, country of origin, patient characteristics, clinical manifestations, diagnostic tools employed, GDA management approaches, treatment effects, in-hospital mortality rates, and any reported complications. Any discrepancy arising during data extraction was resolved through consensus between reviewers (YHL and KTC). If consensus could not be reached, final arbitration was provided by CCH.

Data analysis and group comparisons

The patients in the included studies were categorized into distinct groups to enable comparisons. First, we compared patients who experienced a complicated hospital course (defined as in-hospital mortality or posttreatment complications) with those without such complications. Second, given that pancreatitis is the most prevalent comorbidity associated with GDA, we compared patients with and without pancreatitis.

Statistical analyses were performed using SPSS 15 (SPSS, Chicago, IL, USA). Categorical data are presented as frequencies and percentages, whereas continuous data such as age and GDA size are presented as medians within interquartile ranges (IQRs). The Mann–Whitney U test was employed to assess differences in age and GDA size between groups. For categorical data, intergroup comparisons were performed utilizing the chi-square test.

Search for and identification of studies

A total of 643 citations were initially retrieved from the database search, with an additional 35 citations obtained from the reference lists of relevant studies. Of these, 179 citations were excluded due to duplication, and 150 citations were excluded on the basis of title and abstract screening. This resulted in 349 citations selected for a detailed evaluation. Following a meticulous assessment, 142 articles were further excluded: 27 review articles, 75 case series, and 20 case reports lacking essential patient details. Additionally, seven unpublished studies, conference communications, and animal studies, along with 13 articles that were inaccessible, were excluded from the analysis. Consequently, this meta-analysis included 207 case reports and case series, encompassing data from a total of 224 patients. Figure 1 illustrates the PRISMA flow diagram for the search and identification of included studies.

Characteristics of enrolled patients

Figure 2 depicts the geographic distribution of reported cases. Data originated from patients in 47 countries in all continents, with the United States, India, and Japan constituting 42.0% of all included cases. Of the 224 patients included in this analysis, 221 had documented age data, and the median age was 33.5 years (IQR: 29–39 years). Notably, five case reports lacked information on patient sex. Among the remaining cases, male predominance was observed, with men accounting for 71.2% of the cohort. The majority of patients presented with a comorbidity (85.3%) and had a predisposing factor for GDA (71.9%). These predisposing factors were pancreatitis (Klauß et al., 2012); prior gastrointestinal, biliary, or pancreatic surgery (Moore et al., 2004); percutaneous or internal drainage of abdominal lesion (Basile et al., 2002); intra-abdominal tumor (Androulakakis et al., 2001); peptic ulcer disease (Maggiore, 2001); infectious disease (Sinduja et al., 2021); trauma (Babu et al., 2016); the use of anticoagulants (Harris, Chalhoub & Koirala, 2010); and others (including median arcuate ligament syndrome, systemic lupus erythematosus, hemophilia, and polyarteritis nodosa) (Jensen et al., 2020; Hoorn et al., 2011; Nouira et al., 2006; Dönmez et al., 2005). Data on GDA size were available for 62% of patients, with the median size found to be 3.0 cm (IQR: 2.0–5.0 cm).

Clinical manifestations and diagnostic tools

Abdominal pain (67.0%) was the most prevalent presentation of GDA. Other clinical manifestations were categorized as bleeding in the gastrointestinal tract, abdomen, pancreas, biliary system, or vessels; obstruction in the gastrointestinal tract, pancreatic duct, biliary system, or vessels; and miscellaneous symptoms (Fig. 3). The lesions on the vessels involved either hemorrhage or compression affecting the superior mesenteric vein, portal vein, common hepatic artery, and inferior vena cava. These symptoms could be directly caused by GDA or related to factors associated with GDA.

Abdominal computed tomography scans were found to be the primary diagnostic tool; they were employed in 87.5% of patients and had high diagnostic accuracy of 86.2%. Angiography served a dual role as both a diagnostic tool (employed in 90.2% of patients; accuracy: 100%) and a treatment modality. Abdominal or endoscopic ultrasound was frequently employed for evaluating abdominal lesions, but with low diagnostic accuracy (56.9%). Notably, extraluminal protruding masses and hemorrhage from the ampulla of Vater were identified as endoscopic findings associated with GDA (Giri, Angadi & Bhrugumalla, 2022; Yoshida et al., 2020). GDA-related findings were observed in 23.4% of gastrointestinal endoscopic examinations. Additionally, endoscopic retrograde cholangiopancreatography (9.8%), magnetic resonance imaging (7.6%), and red blood cell scintigraphy (0.9%) were less frequently employed for diagnosis than were other methods. Vascular abnormalities other than GDA were discovered in 19.2% of patients.

GDA management approaches and prognosis

This review included 66 true aneurysm and 158 pseudoaneurysm. Treatment for GDA was conducted emergently in 31.7% of patients, with 34.8% experiencing shock during hospitalization. Transarterial embolization (TAE; 75.0%) and surgery (24.1%) were the primary treatment modalities. Percutaneous thrombin injection (6.7%), vascular stenting (4.5%), and observation (2.7%) were also employed for GDA management. Endovascular managements, including TAE, percutaneous thrombin injection, and vascular stenting, comprised 193 (86.2%) of treatment options. The gastroduodenal artery is part of the collateral network between the celiac trunk and the superior mesenteric artery territories. Therefore, embolization of the GDA in such cases carries the risk of visceral ischemia due to the potential loss of vital collateral blood flow. In this study, we identified one patient with congenital absence of the celiac trunk, one with occlusion of the celiac trunk, three with stenosis of celiac trunk, and one with stenosis of superior mesenteric artery. Among these, the first two patients underwent surgery, while the remaining four underwent TAE. All six patients were successfully treated without sequelae of visceral ischemia.

Regarding prognosis, hemostasis was achieved in 82.6% of patients at the initial treatment attempt. The in-hospital mortality rate was 6.7%, with complications occurring in 16.1% of cases. The mortality rate is higher in patient with true aneurysm (12.1%) than those with pseudoaneurysm (4.3%). Table 1 presents the characteristics, diagnostic tools, treatment strategies, and prognosis of the reviewed cases.

Comparison of the complicated and uncomplicated hospital course groups

Table 2 presents a comparison of patients with complicated versus uncomplicated hospital courses. The two groups had similar characteristics. Regarding clinical manifestations, the complicated hospital course group had higher incidences of bleeding in the peritoneum/retroperitoneum, gastrointestinal obstruction, and biliary obstruction than did the uncomplicated hospital course group (complicated vs. uncomplicated: bleeding to peritoneum and retroperitoneum: 46.5% vs. 26.5%, p = 0.010; gastrointestinal obstruction: 23.3% vs. 8.3%, p = 0.005; biliary obstruction: 37.2% vs. 21.0%, p = 0.025, respectively). Compared with patients in the uncomplicated hospital course group, those with a complicated hospital course underwent fewer TAE procedures, were under observation more frequently, and were more likely to experience shock during hospitalization (complicated vs. uncomplicated: TAE: 62.8% vs. 77.9%, p < 0.001; observation: 11.6% vs. 0.6%, p < 0.001; shock during hospitalization: 51.2% vs. 34.8%, p = 0.012).

Comparison of patients with versus without pancreatitis

We identified 102 (45.5%) patients with pancreatitis, of which 78 had chronic pancreatitis and 24 had acute pancreatitis. Patients with pancreatitis and those without were significantly different in their characteristics and clinical presentations. Patients with pancreatitis tended to be younger, were more likely to be male, and had more comorbidities and predisposing factors. In terms of clinical presentations, abdominal pain, bleeding (gastrointestinal, pancreatic, and biliary), and pancreatic duct and biliary obstructions were all significantly more prevalent in the patients with pancreatitis than in those without. Additionally, thrombin injection was more frequently administered to patients with pancreatitis, and vascular stenting was not applied in this group. Notably, a higher proportion of patients without pancreatitis experienced shock and in-hospital mortality compared with those with pancreatitis. Table 3 presents a detailed comparison of these findings.

Bias

Due to the retrospective study design and heterogeneity of clinical parameters in the included studies, a high risk of bias was noted, which may result in the drawing of weak conclusions.

Strengths and limitations

This meta-analysis systematically reviewed data from 224 patients, derived from 207 case reports spanning 47 countries over a 23-year period. The inclusion of such a substantial number of patients enabled a comprehensive and detailed examination of individuals with GDA. The reviewed cases offer a diverse and global clinical perspective on patients with GDA, potentially offering valuable insights to health-care professionals worldwide.

However, several limitations of this meta-analysis should be acknowledged. First, the significant heterogeneity among the included studies, attributable to their diverse origins, limited our ability to analyze detailed hospital stay data for patients with GDA. Second, international variations in hospital resources and equipment precluded direct comparisons of treatment strategies and outcomes across the reviewed cases. Third, the influence of predisposing factors on the clinical presentations and outcomes of patients with GDA could not be fully elucidated due to the diverse nature of these factors. This heterogeneity may have contributed to distinct clinical courses among patients with GDA resulting from various causes. Finally, it is important to acknowledge the potential for survivorship bias in this review, as most case reports tend to present successfully treated cases.