Primary aorto-enteric fistula (PAEF)is a serious complication of abdominal aortic aneurysm(AAA). We report a patient with PAEF associated with inflammatory AAA who underwent emergent surgery. A 52-year-old male presented with recurrent hematemesis. A computer tomography scan showed a sealed rupture of the AAA adjacent to the duodenum. At surgery, a coin-sized PAEF was noted. The aorta was replaced with a Dacron graft in situ . Histological examination revealed the characteristics of an inflammatory AAA. The postoperative course was uneventful, and there has been no evidence of infection during a follow-up period of 3 years. We discuss the etiologic and surgical considerations regarding this unusual entity.</p

Arata, Takashi

Honjo, Osami

Kurokawa, Tatsuo

Kushida, Yoshio

Matsuno, Tsuyoshi

Yamada, Yukio

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A primary aorto-duodenal fistula associated with an inflammatory abdominal aortic aneurysm: a case report.

Okayama University Scientific Achievement Repository

Acta Medica OkayamaVolume 59, Issue 4 2005 Article 7AUGUST 2005A primary aorto-duodenal fistula associatedwith an inflammatory abdominal aorticaneurysm: a case report.Osami Honjo∗ Yukio Yamada† Takashi Arata‡Tsuyoshi Matsuno∗∗ Tatsuo Kurokawa†† Yoshio Kushida‡‡∗&#xFEFF;Saiseikai Imabari Hospital,†&#xFEFF;Saiseikai Imabari Hospital,‡&#xFEFF;Saiseikai Imabari Hospital,∗∗&#xFEFF;Saiseikai Imabari Hospital,††&#xFEFF;Saiseikai Imabari Hospital,‡‡&#xFEFF;Saiseikai Imabari Hospital,Copyright c©1999 OKAYAMA UNIVERSITY MEDICAL SCHOOL. All rights reserved.A primary aorto-duodenal fistula associatedwith an inflammatory abdominal aorticaneurysm: a case report.∗Osami Honjo, Yukio Yamada, Takashi Arata, Tsuyoshi Matsuno, TatsuoKurokawa, and Yoshio KushidaAbstractPrimary aorto-enteric fistula (PAEF)is a serious complication of abdominal aortic aneurysm(AAA).We report a patient with PAEF associated with inflammatory AAA who underwent emergentsurgery. A 52-year-old male presented with recurrent hematemesis. A computer tomographyscan showed a sealed rupture of the AAA adjacent to the duodenum. At surgery, a coin-sizedPAEF was noted. The aorta was replaced with a Dacron graft in situ . Histological examinationrevealed the characteristics of an inflammatory AAA. The postoperative course was uneventful,and there has been no evidence of infection during a follow-up period of 3 years. We discuss theetiologic and surgical considerations regarding this unusual entity.KEYWORDS: primaryaorto-enteric fistula, inflammatory abdominal aortic aneurysm, rupturedabdominal aortic aneurysm∗PMID: 16155642 [PubMed - indexed for MEDLINE]Copyright (C) OKAYAMA UNIVERSITY MEDICAL SCHOOL A Primary Aorto-duodenal Fistula Associated with an Inﬂammatory Abdominal Aortic Aneurysm:A Case Report Osami Honjo??, Yukio Yamada?, Takashi Arata?, Tsuyoshi Matsuno?,Tatsuo Kurokawa?, and Yoshio Kushida??Departments of Cardiovascular Surgery, ?Surgery and?Pathology,Saiseikai Imabari Hospital, Imabari, Ehime 799-1592, Japan Primary aorto-enteric ﬁstula(PAEF)is a serious complication of abdominal aortic aneurysm(AAA).We report a patient with PAEF associated with inﬂammatory AAA who underwent emergent surgery. A 52-year-old male presented with recurrent hematemesis. A computer tomography scan showed a sealed rupture of the AAA adjacent to the duodenum. At surgery, a coin-sized PAEF was noted. The aorta was replaced with a Dacron graft . Histological examination revealed the characteristics of an inﬂammatory AAA. The postoperative course was uneventful, and there has been no evidence of infection during a follow-up period of 3 years. We discuss the etiologic and surgical considerations regarding this unusual entity.Key words:primary aorto-enteric ﬁstula, inﬂammatory abdominal aortic aneurysm, ruptured abdominal aortic aneurysm P rimary aorto-enteric ﬁstula(PAEF)is a rare but serious complication of abdominal aortic aneurysm(AAA), and the outcome of surgical treatment is poor［1,2］. The surgical results depend on preoperative condition of the patient and degree of contamination of the operative ﬁeld［1, 2］. Inﬂammatory AAA is one of the causes of PAEF, and a few cases of PAEF caused by inﬂammatory AAA have been reported［3, 4］. We report here the successful treatment of a case of PAEF associated with inﬂammatory AAA and discuss the etiologic and surgical considerations regarding this un-usual entity.Case Report A 52-year-old male was referred to our institution for treatment of recurrent hematemesis and melena. He had had hematemesis and melena 2 weeks before admission and had visited a local hospital. Emergency exploratory laparotomy was performed due to uncontrollable hematemesis, but no obvious cause in the abdomen was found. A repeat endoscopic examination showed an extrinsic pulsating mass protruding into the internal lumen of the second portion of the duodenum. The recurrent gastrointestinal bleeding was uncontrollable, and the patient was transferred to our institution. An abdominal aortic aneurysm of 60 mm in diameter was noted on a computer tomography(CT)scan, which was adjacent to the third portion of the duodenum(Fig. 1). Abdominal aortography revealed a saccular-shaped AAA located approximately 30 mm distal of the renal arteries(Fig. 2).At emergent surgery, the aneurysm appeared to be Received May 27,2004;accepted March 23,2005.?Corresponding author.Phone:＋81-86-235-7359 Fax:＋81-86-235-7431 E-mail:Osami Honjo＠sickkids.ca(O.Honjo)http://www.lib.okayama-u.ac.jp/www/acta/Acta Med. Okayama, 2005 Vol. 59 , No. 4, pp. 161-164 Case Report Copyright?ｃ2005by Okayama University Medical School.1Honjo et al.: A primary aorto-duodenal fistula associated with anProduced by The Berkeley Electronic Press, 2005inﬂammatory, and the right lateral wall of the aneurysm had a communication with the third portion of the duode-num. Inspection of the internal lumen of the aorta showed a coin-sized aorto-duodenal ﬁstula(Fig. 3). The markedly thickened arterial wall was resected to the extent that was possible. The aorta was replaced with a Dacron Y graft in situ. The perforated region of the duodenum was resected and was anastomosed in an end-to-end fashion.The cavity was irrigated with 10 liters of saline. The omentum was placed between the aorta and duodenum.A culture of the aneurysmal wall showed the presence of Candida species, and an antifungal agent was administer-ed for 2 months. The results of histological examination showed that chronic inﬂammatory cells had focally inﬁltrated into the aneurysmal wall (Fig. 4), and that neutrophilic inﬁltration with abscess formation was seen around the ﬁstula. The patient’s postoperative course was uneventful, and there has been no evidence of infection during a follow-up period of 3 years.Discussion PAEF, direct communication between the aorta and the intestinal tract, is mainly due to an atherosclerotic aneurysm, and other causes include mycotic aneurysm,radiation trauma, and inﬂammatory AAA［1, 2］.Although 5 of infrarenal AAAs have the characteristics of inﬂammatory AAA, PAEFs resulting  from inﬂammatory AAA are extremely rare［3, 4］. Ikeda et al. reported a 50-year-old woman who had PAEF associated with multiple inﬂammatory AAAs［3］, and Mii et al. reported a 28-year-old man who had primary aorto-jejunal ﬁstula associated with inﬂammatory AAA［4］. Together with the present case, the clinical charac-teristics of those patients include relatively young age and the presence of a rapidly developed saccular-shaped aneur-ysm. Interestingly, there were no symptoms associated with systemic inﬂammation such as high-grade fever,Honjo et al. Acta Med. Okayama Vol. 59 , No. 4 162 Fig.1  A CT scan showing the sealed rupture of the AAA adjacent to the third portion of the duodenum.Fig. 2  Aortography revealing a saccular-shaped AAA with no extravasation.Fig.3  Intraoperative ﬁnding of a coin-sized aorto-duodenal ﬁstula.The arrow shows the ﬁstularization.2Acta Medica Okayama, Vol. 59 [2005], Iss. 4, Art. 7http://escholarship.lib.okayama-u.ac.jp/amo/vol59/iss4/7despite the fact that all 3 patients had elevated white blood cells and C-reactive protein. The early to medium term results of those patients were encouraging, even though the overall surgical results in patients with PAEF have been poor. Such cases must receive careful follow-up,because the native aortic tissue in the anastomotic sites still has the potential of recurrent inﬂammation and subse-quent ﬁstulization.It is controversial whether the chronic inﬂammation of the aneurysmal wall results purely from the inﬂammatory process, i.e., a so-called “non-speciﬁc inﬂammatory aneurysm,”or from infection, i.e., a mycotic aneurysm.In the present case, a culture of the aneurysmal wall showed the presence of Candida species;however, the patient had no symptoms of active infection, and there was also no evidence of endocarditis or other focuses.Furthermore, bacteriological study of mycotic aneurysm［5］showed that the common organisms cultured from aneurismal walls included Staphylococcus aureus and Salmonella, and mycotic aneurysm due to Candida species was extremely rare［5］. In addition, the charac-teristic CT ﬁndings in mycotic aneurysms, which include the sudden appearance of an aneurysm on a previously normal aorta in a febrile patient, a soft tissue mass surrounding the aneurysm, and an adjacent vertebral osteomyelitis［6］, were not fully correlated with ﬁndings in present case. The clinical and pathological ﬁndings in our case indicated that Candida species might not be a primary cause of inﬂammatory aneurysm but might be a secondary contamination through the ﬁstula, and we concluded that it was a non-speciﬁc inﬂammatory aneur-ysm rather than a mycotic aneurysm.Despite the presence of contamination,most surgeons have reconstructed diseased aortae with in situ grafts［1-4］. Extra-anatomic bypass grafting is an alternative approach for treatment of PAEF to avoid the use of a prosthetic graft in the contaminated region［7］. How-ever, a high mortality rate has been reported in patients treated with extra-anatomic bypass, since not only does the procedure involve high risks of limb loss and aortic stump blowout, but also it is often performed in severely ill patients with gross infection in the operative ﬁeld. The selection of surgical approach depends on the presence of sepsis and the degree of contamination. If a severe purulent infection is present in periaortic tissue, extra-anatomic bypass should be selected. Our patient was treated with an in situ graft because the contamination of the operative ﬁeld was localized. Careful irrigation of the operative ﬁeld is mandatory regardless of the presence or absence of gross contamination, and the proximal end of the prosthetic graft should be wrapped with the omentum to prevent recurrent ﬁstulization, especially in cases of inﬂammatory aneurysm.PAEF with Inﬂammatory AAA August 2005 Fig.4  Histological ﬁndings of the aneurysmal wall:chronic inﬂammatory cells inﬁltrated into the aneurysmal wall(A), and severe chronic inﬂammatory cell inﬁltration and localized neutrophilic inﬁltration with abscess formation(B).A  B 1633Honjo et al.: A primary aorto-duodenal fistula associated with anProduced by The Berkeley Electronic Press, 2005 References 1. Sweeney MS and Gadacz TR:Primary aortoduodenal ﬁstula:Manifes-tation, diagnosis, and treatment. Surgery(1984)96:492-497.2. Tareen AH and Schroeder TV:Primary aortoenteric ﬁstula:Two new case reports and review of 44 previously reported cases. Eur J Vasc Endovasc Surg (1996)12:5-10.3. Ikeda K, Abe T, Itou M, Tamiya Y, Tanaka T and Kazui T:Successful surgical treatment of primary aorto-duodenal ﬁstula associated with inﬂammatory abdominal aortic aneurysm:A case report. Ann Thorac Cardiovasc Surg (1999)5:194-197.4. Mii S, Onohara T, Okadome K, Fukuda A and Sugimachi K:Surgical repair of primary aorto-jejunal ﬁstula associated with non-speciﬁc inﬂammatory abdominal aortic aneurysm. Eur J Vasc Surg (1991)5:355-357.5. Brown SL, Busuttil RW, Baker JD, Machleder HL, Moore WS and Barker WF:Bacterioligic and surgical determinants of survival in patients with mycotic aneurysms. J Vasc Surg (1984)1:541-547.6. Parellada JA, Palmer J, Monill JM, Zidan A, Gimenez AM and Moreno A:Mycotic aneurysm of the abdominal aorta:CT ﬁndings in three patients. Abdom Imaging (1997)22:321-324.7. Pagni S, Denatale RW, Sweeney T, McLauglin C and Ferneini AM:Primary aorto-duodenal ﬁstula secondary to infected abdominal aortic aneurysm:the role of local debridement and extra-anatomic bypass. J Cardiovasc Surg (1999)40:31-35.Honjo et al. Acta Med. Okayama Vol. 59 , No. 4 1644Acta Medica Okayama, Vol. 59 [2005], Iss. 4, Art. 7http://escholarship.lib.okayama-u.ac.jp/amo/vol59/iss4/7

A primary aorto-duodenal fistula associated with an inflammatory abdominal aortic aneurysm: a case report.

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