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Case Report
A Rare Cause of Primary Aortoenteric Fistula:
Streptococcus parasanguinis Aortitis
Fredy Nehme, Kyle Rowe, Cyrus Munguti, and Imad Nassif
Department of Internal Medicine, Kansas University School of Medicine, Wichita, KS, USA
Correspondence should be addressed to Fredy Nehme; nehme.fredy@gmail.com
Received November ; Revised January ; Accepted January ; Published January
Academic Editor: I. Michael Leitman
Copyright © Fredy Nehme et al. is is an open access article distributed under the Creative Commons Attribution License,
which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
Primary aortoenteric stula is a rare cause of upper gastrointestinal bleed but can lead to signicant mortality if the diagnosis is
delayed. Aortitis, characterized by inammation of the aortic wall, is a rare cause of aortoenteric stula. We present a case report of
a -year-old male patient with infectious aortoenteric stula secondary to Streptococcus parasanguinis,alongwithareviewofthe
literature. is case demonstrates the importance of early diagnosis and aggressive surgical treatment of aortoenteric stulae and
recognizing infectious aortitis as a potential etiology.
1. Introduction
Primary aortoenteric stula (PAEF) is a direct communi-
cation between the aorta and the intestinal lumen without
prior reconstructive procedures on the abdominal aorta.
Although rare with an estimated prevalence of .% [],
PAEF is a severe life threatening emergency that should
be promptly recognized. Urgent surgical repair is usually
warranted. PAEF commonly predominates in the third and
fourth portion of the duodenum []. e leading causes of
PAEF are atherosclerosis and aortic aneurysm in about %
of cases, while aortitis remains a less common etiology [].
In this report, we describe a rare case of PAEF caused by
infective aortitis due to Streptococcus parasanguinis.Early
recognition and urgent surgical repair lead to a favorable
outcome.
2. Case Presentation
A -year-old male presented with chief complains of acute
onset abdominal pain, back pain, and hematemesis. e
patient reported vomiting around ounces of blood followed
byanepisodeofmelena.Hereportedchills,fatigue,diaphore-
sis, and subjective fevers for the last week. He denied chest
pain,shortnessofbreath,orcough.Hispastmedicalhistory
was signicant for a right lower lobe stage IB non-small
cell lung cancer treated with radiation therapy currently in
remission and polyangiitis obliterans with resultant upper
and lower limb amputations. Physical examination revealed
a temperature of . F, with a regular pulse rate of /min
and a blood pressure of /mmHg. Abdominal exam
elicited mild diuse tenderness without guarding. Laboratory
investigation was signicant for anemia with hemoglobin
of g/dL. Inammatory markers were elevated with a C-
reactive protein of . mg/dL and erythrocyte sedimentation
rate of mm/h. No coagulopathy or thrombocytopenia
was noted. Blood cultures were not drawn. Esophagogas-
troduodenoscopy (EGD) revealed a large pulsating bulge in
the second portion of the duodenum. A mm ulcer with
an adherent clot was also visualized in the same region
withoutactivebleeding(Figure).Urgentcomputedtomog-
raphy angiography (CTA) showed a . cm aortic aneurysm,
marked so tissue thickening of the aortic wall, and a small
pocket of gas within the so tissue of the aorta near the
second portion of the duodenum (Figure ). With nd-
ings suspicious of aortitis and aortoenteric stula, vascular
surgery was consulted and urgent exploratory laparotomy
was scheduled. Upon exploration, adhesions between the
small bowel, the omentum, and the anterior abdominal wall
were carefully taken down. ere was clear evidence of a
stula with adherence of the small bowel to the anterior
lateral aneurysm wall. e aneurysmal portion involving the
Hindawi
Case Reports in Gastrointestinal Medicine
Volume 2017, Article ID 9087308, 3 pages
https://doi.org/10.1155/2017/9087308
Case Reports in Gastrointestinal Medicine
F : EGD showi ng a large pulsating bulge in the se cond portion
of the duodenum with an adjacent mm ulcer.
F : CTA showing a . cm aortic aneurysm, marked so tissue
thickening of the aortic wall, and a small pocket of gas within the so
tissue of the aorta (so tissue gas designated by the arrow).
stula was noted to contain signicant purulence requiring
surgical debridement. e involved aortic aneurysm was
resected. A -mm Hemashield tube gra was sewn between
the infrarenal aorta and distal aorta with - Prolene sutures.
A duodenojejunostomy with resection of the involved fourth
portion of the duodenum was then performed.
A mycotic aneurysm was strongly suspected and the
patient was started empirically on Meropenem. Cultures
from the excised aortic aneurysm were positive for Strepto-
coccus parasanguinis and antibiotics were deescalated appro-
priately. Acid-fast bacilli and fungal stains were negative.
Surgicalpathologyrevealedintensechronicactiveinamma-
tion with the presence of necrotizing granulomas (Figure ).
Resection margins were free of active inammation. A -
week course of antibiotics was completed with signicant
improvement. Follow-up at months demonstrated complete
resolution of symptoms.
3. Discussion
Aortoenteric stula (AEF), dened as an abnormal connec-
tion between the aorta and the gastrointestinal tract, is a
rare cause of upper gastrointestinal bleeding but can lead to
signicant morbidity and mortality if not promptly recog-
nized. Two types of AEFs are described, primary aortoenteric
stula arising de novo between the aorta and the bowel or
F : Surgical pathology revealing intense active inammation
with the presence of necrotizing granulomas.
secondary aortoenteric stula (SAEF) occurring aer aortic
reconstruction surgery. SAEF is far more common [].
e “herald bleed” is a common presentation of AEF
that needs to be recognized early in order to prevent a later
catastrophic hemorrhage. e diagnosis of PAEF is elusive
as the classic triad of gastrointestinal bleeding, abdominal
pain, and palpable mass is only found in % of patients []
and upper gastrointestinal endoscopy has a sensitivity of only
% []. It is important to recognize the endoscopic ndings
suggestiveofAEF,particularlythepresenceofapulsating
mass, as dislodging a fresh thrombus may lead to massive
hemorrhage. CTA is currently the rst line imaging modality
to evaluate an AEF bleed [].
Aortitis, characterized by inammation of the aortic
wall [], is an uncommon cause of aortic aneurysm with
inammatory aneurysms compromising only to % of
abdominal aortic aneurysms []. e most common causes of
aortitis include the large vessel vasculitis, giant cell arteritis,
and Takayasu arteritis. Other rheumatologic diseases are
also associated with aortitis []. Infectious aortitis is a rare
but life threatening condition that must be dierentiated
from other inammatory conditions as treatment strategies
diverge widely []. Bacterial seeding of the aortic wall,
usually with an underlying pathology, occurs via the vasa
vasorum[].isresultsintheformationofananeurysmor
pseudoaneurysm that erodes into the adjacent structures [].
In our case, the diagnosis of aortitis was strongly supported
by elevated inammatory markers, CTA ndings of marked
so tissue thickening of the aortic wall, and pathology results
demonstrating intense active inammation with the presence
of necrotizing granulomas. e intraoperative nding of
signicant purulent material around the involved aortic
segment, the presence of periaortic gas on CTA, and the
isolation of a bacterial organism were in favor of an infectious
etiology which prompted the initiation of broad spectrum
antibiotics. However, the presence of periaortic gas can also
be attributed to presence of an AEF. Although Streptococcus
parasanguinis, amemberoftheviridansgroup,isassociated
with a variety of infections, mycotic aneurysm is uncommon.
In our case, the source of infection was unknown.
erapy of infected aneurysms relies mainly on a com-
bination of antibiotic therapy, surgical debridement, and, if
Case Reports in Gastrointestinal Medicine
possible, revascularization []. Timing and type of surgical
repair depend on multiple factors including severity of the
clinical presentation, patient comorbidities and type of AEF.
Open surgical repair generally involves vascular control of the
aorta followed by debridement of all infected and necrotic
tissue, repair or resection of the intestinal defect, and in
situ aortic reconstruction with a prosthetic gra []. While
revascularization is usually performed immediately in PAEF,
the timing and type of revascularization are debated in SAEF.
For patients who are not considered good surgical candidates
for open repair, an endovascular approach is suggested either
as a bridge procedure to denitive repair [] or as an adjunct
to palliative treatment []. Options include endovascular bal-
loon occlusion of the aorta, endovascular coil embolization,
and stent-gra repair []. Optimal antibiotic duration is
uncertain and varies depending on several factors. Usually,
six weeks of antibacterial therapy is suggested [].
4. Conclusion
Early diagnosis and aggressive surgical treatment of an AEF
are vital to achieve a positive outcome. Aortitis is a rare cause
ofAEFthatshouldbeconsideredbasedonclinical,imaging,
and surgical ndings. Infectious aortitis, as seen in this case,
should be considered as an etiology of aortitis in cases with
suggestive history and radiological ndings.
Competing Interests
is statement is to certify that all authors have no conict of
interests to declare.
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