Secondary aortoenteric fistula: a narrative review of the view of the surgeon

Abstract

Aortoenteric fistula (AEF) is defined as the abnormal communication between the aorta and the gastrointestinal tract. AEFs are divided into primary and secondary usually after abdominal aortic aneurysm (AAA) recovery and are a rare but quite dangerous cause of gastrointestinal bleeding that the general surgeon may face during his/her career. Secondary AEF was first described in 1953 to a 44-year-old woman 3 months after an AAA operation. This review presents the role of the surgeon in the management of secondary aortoenteric fistulas. AEFs are a rare but fatal gastrointestinal bleeding cause that the general surgeon may be asked to manage. Diagnosis requires the combination of strong clinical suspicion and the presence of a history of AAA surgery. Although a vascular surgery case, general surgeons play a role in choosing the technique of restoring the intestinal tract, which seems to be significantly related to subsequent morbidity and mortality.

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Aorta
Review paper
Corresponding author:
Dr. Francesk Mulita
Department of Surgery
General University
Hospital of Patras
Patras, Greece
E-mail: oknarfmulita@
hotmail.com
1Department of Surgery, General University Hospital of Patras, Patras, Greece
2
Department of Cardiothoracic and Vascular Surgery, WestpfalzKlinikum,
Kaiserslautern, Germany
3
Department of Vascular Surgery, General University Hospital of Patras, Patras, Greece
4
Department of Interventional Radiology, General University Hospital of Patras,
Patras, Greece
5Health Centre of Akrata, Akrata, Greece
6
Department of Cardiothoracic Surgery, General University Hospital of Patras, Patras,
Greece
Submitted: 31 January 2024; Accepted: 21 March 2024
Online publication: 30 March 2024
Arch Med Sci Atheroscler Dis 2024; 9; e66–e71
DOI: https://doi.org/10.5114/amsad/186358
Copyright © 2024 Termedia & Banach
Secondary aortoenteric fistula: anarrative review
of the view of the surgeon
Francesk Mulita1, Vasileios Leivaditis2, Georgios-Ioannis Verras1, Christos Pitros3,
Platon Dimopoulos4, Paraskevi F. Katsakiori5, Danai Dafnomili1, Levan Tchabashvili1, Konstantinos
Tasios1, Dimitrios Kehagias1, Andreas Antzoulas1, Spyros Papadoulas3, Efstratios Koletsis6
Abstract
Aortoenteric fistula (AEF) is defined as the abnormal communication be-
tween the aorta and the gastrointestinal tract. AEFs are divided into primary
and secondary usually after abdominal aortic aneurysm (AAA) recovery and
are arare but quite dangerous cause of gastrointestinal bleeding that the
general surgeon may face during his/her career. Secondary AEF was first
described in 1953 to a44-year-old woman 3 months after an AAA operation.
This review presents the role of the surgeon in the management of second-
ary aortoenteric fistulas. AEFs are arare but fatal gastrointestinal bleeding
cause that the general surgeon may be asked to manage. Diagnosis requires
the combination of strong clinical suspicion and the presence of ahistory of
AAA surgery. Although avascular surgery case, general surgeons play arole
in choosing the technique of restoring the intestinal tract, which seems to
be significantly related to subsequent morbidity and mortality.
Key words: aortoenteric fistula, abdominal aortic aneurysm, aorta,
secondary.
Introduction
Aortoenteric fistulas were first described by Cooper [1] as acompli-
cation of abdominal aortic aneurysms, while the first case is described
in 1843 by Salmon [2] and defined as the abnormal communication
between the aorta and the gastrointestinal tract. Aortoenteric fistulas
are divided into primary and secondary usually after abdominal aortic
aneurysm recovery and are arare but quite dangerous cause of gastro-
intestinal bleeding that the general surgeon may face during his/her ca-
reer. Secondary aortoenteric fistula (SAEF) was first described by Brock in
1953 in the case of a44-year-old woman 3 months after AAA operation
[3]. This review presents the role of the surgeon in the management of
secondary aortoenteric fistulas.

Secondary aortoenteric fistula: anarrative review of the view of the surgeon
Arch Med Sci Atheroscler Dis 2024 e67
The aim of our review is to provide abrief over-
view of arare but potentially rapidly fatal condition
that general surgeons worldwide might be called
to face in the emergency setting. Although special-
ised management of this condition is paramount
to the effective treatment of the SAEF, emergency
presentations often require management and sur-
gically-led stabilisation from the general surgeon
that is not trained in vascular surgery.
We summarised, in aconcise way, the knowl-
edge deriving from the available literature about
SAEFs, providing the surgeon’s view.
Material and methods
Studies were identified by searching electronic
databases and scanning bibliographic references of
articles. The National Library of Medicine’s Medline
database was searched using the PubMed inter-
face and Scopus from 1990 to 2023. No language
constraints were used. The last search was run on
12 December 2023. Keywords were selected us-
ing medical subject headings (MeSH) for PubMed
and MeSH/Emtree for Scopus. The keywords were
“secondary fistula”, “aortoenteric”, “aortojejunal”,
and “aortoiliac”. The databases were searched with
an unrestricted search strategy, applying exploded
MeSH and keywords combined with the Boolean
operators AND or OR to retrieve relevant reports as
reported in Table I. Asecond-level search included
amanual screen of the reference lists of the articles
identified through the electronic search. Eligibility
assessment was performed independently in an
unblinded, standardised manner by 2 reviewers;
disagreements between reviewers were resolved
by consensus (Figure 1).
Results
Epidemiology
SAEFs can occur after both open and intravas-
cular reconstructive surgery for an aortic aneu-
rysm or other aortic diseases with incidence rang-
Figure 1. PRISMA Flow diagram. Alist of included articles
Table I. The database search strategy
Search strategy Results
Medline (PubMed)
1((«neoplasm metastasis»[MeSH Terms] OR («neoplasm»[All Fields] AND «metastasis»[All Fields])
OR «neoplasm metastasis»[All Fields] OR «secondaries»[All Fields] OR «secondary»[MeSH
Subheading] OR «secondary»[All Fields]) AND («fistula»[MeSH Terms] OR «fistula»[All Fields] OR
«fistulas»[All Fields] OR «fistula s»[All Fields] OR «fistulae»[All Fields] OR «fistulaes»[All Fields])
10 727
2(«aortoenteric»[All Fields] OR «aortojejunal»[All Fields] OR «aortocolonic»[All Fields]) 937
3#1 AND #2 407
Scopus
TITLE-ABS-KEY: secondary fistula AND (aortoenteric OR aortojejunal OR aortocolonic) 102
Identification of studies via databases and registers
Records identified from:
Medline (n = 308)
Scopus (n = 93)
Records screened (n = 329)
Reports sought for retrieval (n = 57)
Reports assessed for eligibility (n = 57)
Studies included in results (n = 37)
Records removed before screening:
Duplicate records removed (n = 72)
Records excluded as irrelevant (n = 272)
Reports not retrieved (n = 0)
Reports excluded:
Primary aortoenteric fistula (n = 7)
Review (n = 13)
IdentificationIncluded Screening

Francesk Mulita, Vasileios Leivaditis, Georgios-Ioannis Verras, Christos Pitros, Platon Dimopoulos, Paraskevi F. Katsakiori, Danai Dafnomili,
Levan Tchabashvili, Konstantinos Tasios, Dimitrios Kehagias, Andreas Antzoulas, Spyros Papadoulas, Efstratios Koletsis
e68 Arch Med Sci Atheroscler Dis 2024
ing from 0.36 to 1.6% and increasing to 12–33%
in cases who have an infected graft [4, 5]. They
are more common in adults, and the proportion
of male to female is 8 : 1 [6, 7]. They usually oc-
cur within the first year after surgery (time range
1–20 years) [7, 8]. The mortality rate of SAEF has
been reported to be up to 50–70% [9].
A recent series [10] demonstrated that SAEF
continues to be alife-threatening condition, with
a 27% in-hospital mortality rate, which is sub-
stantially higher upon emergency presentation,
despite prompt diagnosis and treatment. The ma-
jority of fistulas involving the intestinal tract are
between the aorta and the 3rd–4th portion of the
duodenum (between the abdominal aorta and the
superior mesenteric artery) in 74% of cases, fol-
lowed by the small intestine (jejunum more com-
monly than the ileum) 19%, the colon 5%, and the
appendix 1% [11–14].
Pathogenesis
The pathogenesis of SAEFs is not entirely clear al-
though some hypotheses have been described. The
main mechanism seems to be the gradual erosion
of the aortic graft in the intestine, with or without
the presence of inflammation from the intestinal
contents. The constant irritation of the intestinal
wall by the aortic pulsation seems to play an im-
portant role, which explains why most fistulas ap-
pear at the proximal anastomotic site of the third
portion of the duodenum, which is pressed between
the aorta and the superior mesenteric artery in the
retroperitoneal space [3, 15–17]. Other hypotheses
presuppose the presence of local inflammation and
subsequent erosion of the intestinal wall due to in-
fection of the aortic graft during the first operation
[11, 15, 18–20]. Apositive preoperative blood cul-
ture predicts apoor outcome [21, 22].
Clinical picture – diagnosis
The diagnosis of an aortoenteric fistula can
become quite challenging, and strong clinical sus-
picion is required. Although the clinical picture is
directly related to the part of the gastrointestinal
tract where the fistula has formed, typically pa-
tients come with at least one of the following:
gastrointestinal bleeding (melena, haematoche-
zia, haematemesis) 80%, sepsis (44%), abdominal
pain (30%), lumbar pain 15%, groin mass (12%),
and abdominal pulsatile mass (6%). Other under-
reported symptoms include acute limb ischaemia,
pain, vomiting, ileus, and septic emboli to the legs
[3, 5, 13]. The typical triad: gastrointestinal bleed-
ing, abdominal pain, and palpable mass, is found
in only 6–12% of cases [23]. Intermittent bleeding
followed by massive bleeding (Herald bleeding)
is typical for the disease and raises astrong sus-
picion of diagnosis although it occurs in approx-
imately 60% [5, 18, 20, 24, 25]. Song et al. [26]
proposed that the occurrence of herald bleeding
had amean frequency of 3.6 episodes. They also
found that the time interval between the first her-
ald bleeding and massive exsanguination varied
from 5 h to 5 months, with amedian of 4 days.
More than 50% of cases had a duration longer
than 3 days [26]. This time frame can be used as
an opportunity for the treatment of fistulas [14].
For imaging, several methods have been pro-
posed, the most useful being oesophagogastro-
duodenoscopy and computed tomography (CT).
The choice of imaging method depends on the pa-
tient’s condition, the presence or not of bleeding,
and its severity. In the case of massive bleeding,
the patient should be taken directly to the oper-
ating room without further delay. In acute bleed-
ing in stable patients, endoscopy is usually per-
formed but with low accuracy in the diagnosis of
Figure 2. A– CT angiography showing the intravenous contrast extravasation in the lumen of the duodenum (red
arrow). B – The migrated duodenal stent is also recognised (red arrow)
A B

Secondary aortoenteric fistula: anarrative review of the view of the surgeon
Arch Med Sci Atheroscler Dis 2024 e69
AEF (25–40%) [6, 26, 27]. As already mentioned,
most aortoenteric fistulas are found in the 3rd-4th
portion of the duodenum and for this reason the
endoscope should reach the entire length of the
duodenum, while use of an enteroscopy or pae-
diatric endoscopes is recommended [6, 8, 28–30].
The presence of fresh blood and clots are indica-
tive. Colonoscopy is not often cited as adiagnostic
method [31, 32].
Spiral CT is fast and easily accessible from most
centres. Intravenous contrast-enhanced CT helps
both in the diagnosis and in the later decision of
surgical repair of the aorta due to the anatomical
information it provides [25, 33]. CT findings sug-
gestive of AEF are air around the aorta, bowel wall
oedema around the aorta, and the loss of afatty
plane between the aorta and GI tract (Figure 2).
The fistula itself may not be visualised. CT angi-
ography can be negative, despite an active bleed
from an AEF [32]. Despite the great value of imag-
ing, the clinical presence of gastrointestinal bleed-
ing is very important for differentiating AEFs from
other entities such as perigraft infection without
fistulation.
Treatment
The treatment of SAEFs, regardless of their lo-
cation, is exclusively surgical, and without it the
mortality is 100%. With appropriate treatment,
survival rates greatly varying between 18% and
93% have been reported [22, 34]. The choice of
technique depends on the patient’s condition, the
severity of bleeding, comorbidities, and the pres-
ence or absence of inflammation. In the first phase,
resuscitation and haemodynamic support are of
priority. Preoperative intravenous broad-spectrum
antibiotic therapy for Gram+, Gram–, and enteric
pathogens may be administrated followed by tar-
geted therapy [35].
The treatment of AEF includes graft excision
and extra-anatomic bypass or in situ graft replace-
ment and simple graft excision alone. Conserva-
tive or palliative treatment, utilising antibiotics
and drainage or irrigation, is also acceptable for
patients unfit for surgery [36]. Endovascular repair
is aless invasive alternative, including limited re-
ports for the treatment of AEFs. In SAEF, endovas-
cular treatment (Figure 3) is performed as abridg-
ing therapy to open repair to stabilise the patient
until open repair is accessible. Arecent multi-cen-
tre study [37] for the management of SAEFs
found that there was no discernible distinction
in all-cause mortality rates among patients who
received EVAR or OAR as initial therapy for SAEF.
In the acute setting, EVAR may be considered as
aprimary treatment or abridge to definitive OAR
in conjunction with broad-spectrum antimicrobial
therapy for patients with SAEF. Kakkos et al. [13]
came to conclusion that for SAEFs, endovascular
surgery is associated with ahigher early survival
rate (p < 0.001) than open surgery, when appropri-
ate, since in hospital mortality was 7.1% for endo-
vascular surgery and 33.9% for open repair. Long-
term follow-up erodes the majority of this benefit,
because late sepsis occurred more often after en-
dovascular surgery (2-year rate 42% vs. 19% for
open, p = 0.001), suggesting that in certain pa-
tients, astaged approach with early conversion to
in situ vein grafting may produce the best results.
Open procedure (staged or not staged) has as its
Figure 3. A, B – Extravasation of the IV contrast during the aortography and occlusion after the aortic stent place-
ment (black arrows). The migrated duodenal stent is also recognised
A B

Francesk Mulita, Vasileios Leivaditis, Georgios-Ioannis Verras, Christos Pitros, Platon Dimopoulos, Paraskevi F. Katsakiori, Danai Dafnomili,
Levan Tchabashvili, Konstantinos Tasios, Dimitrios Kehagias, Andreas Antzoulas, Spyros Papadoulas, Efstratios Koletsis
e70 Arch Med Sci Atheroscler Dis 2024
fundamental objectives the confirmation of the
diagnosis, the control of bleeding and inflamma-
tion, and the restoration of the continuity of the
intestinal tract. Older series demonstrate opera-
tive mortality as high as 64% [38]. Arecent study
[39] provides the 20-year experience of asingle
centre, reporting significant improvement in 30-
day (p = 0.03) and 90-day (p = 0.008) mortality
even on risk-adjusted analysis, with no significant
difference in 1-year survival overall.
The management of aortoduodenal fistulas
has been studied more than the other subtypes
of aortoenteric fistulas due to the frequency of oc-
currence. Initially, alaparotomy is conducted with
temporary in situ revascularisation with aprosthet-
ic graft, and later, astaged extra-anatomic bypass
followed by transabdominal removal of the tem-
porarily placed graft. Duodenal exclusion is not an
obligatory adjunct to duodenal repairs, but in some
cases resection of the fistula-bearing area of the
aneurysm resected en bloc with the attached du-
odenum may be needed. In some cases extensive
destruction of the duodenal wall is noted, and then
pylorus-preserving pancreatoduodenectomy is an
option. Avascularised pedicle of the greater omen-
tum can be used to wrap around the prosthetic
graft or cover the infected surgical field for the pur-
pose of preventing agraft infection. Aortojejunal
fistula are rare and usually treated with primary
repair, and the jejunal defect is closed using alin-
ear stapler [40, 41]. Aortocolonic fistulas are better
treated with some type of colectomy and/ or trans-
verse colostomy. In general, patients who undergo
bowel resection have aworse prognosis than those
who have simple repair [42]. Iliac-appendiceal fis-
tula is arare medical entit, which is treated with
appendectomy and partial cecectomy [43].
Limitations: As a narrative review, our study
has inherent limitations. Our study was done in
a systematic way, but it is not asystematic re-
view, and asignificant risk of bias remains. Many
of the data we have presented may derive from
low-quality studies such us case reports. No sta-
tistical analysis was done to further interpret the
collected data.
In conclusion, the role of the general surgeon
in managing an aortoenteric fistula (AEF) is crucial
and multifaceted. In short, it can be summarised
in the points below:
• The surgeon must confirm the diagnosis of
SAEF. This is often done through imaging tech-
niques like computed tomography (CT), which
provides superior images compared to other
diagnostic modalities.
• One of the primary responsibilities of the sur-
geon is to control the bleeding associated with
SAEF. This is particularly important because
most AEFs are heralded by repetitive gastroin-
testinal bleeds
• The surgeon must repair the damaged area of
the bowel
• If the condition is caused by an AAA or aprevi-
ous aortic graft, the surgeon will need to remove
the aneurysm or graft that is causing the issue
• Once SAEF is diagnosed, preventive measures,
such as antibiotic therapy, delicate surgery for
eradication of septic focus with thorough de-
bridement of infected and devitalised tissue,
and reconstruction of the excised aorta by ex-
tra-anatomic or an in situ route, are required
• Over time, operative mortality rates have de-
creased, possibly due to improvements in
perioperative care and the advent of endovas-
cular techniques
Aortoenteric fistulas are arare but fatal gas-
trointestinal bleeding cause that the general
surgeon may be asked to manage. Diagnosis
requires the combination of strong clinical sus-
picion and the presence of ahistory of AAA sur-
gery. Although avascular surgery case, general
surgeons play arole in choosing the technique
of restoring the intestinal tract, which seems to
be significantly related to subsequent morbidity
and mortality.
Conflict of interest
The authors declare no conflict of interest.
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