Primary Aortoenteric Fistula Due to Septic Aortitis

Georgios Skourtis, Papacharalambous Gerasimos, Makris Sotirios, Kaskis Fotios, Kastrisios Georgios, Goulas Sotirios, Antoniou Ioannis, Giannakakis Sotirios, and Maltezos Chrisostomos, Athens, Greece

Primary aortoenteric stula is most commonly caused from erosion of the bowel wall by an abdominal aortic aneurysm. Septic aortitis with pseudoaneurysm formation and nally erosion into the duodenum represents a rare cause that has been described in very few patients in the literature. We present a rare clinical case of Salmonella aortitis and associated infrarenal aortic pseudoaneurysm that evolved into an aortoduodenal stula. A 51-year-old man was admitted in our hospital with symptoms and signs of sepsis caused by Salmonella bacteremia. Imaging studies revealed an infrarenal aortic pseudoaneurysm. The patient presented hemodynamic instability, and during emergency laparotomy a stula was found between the third portion of the duodenum and a false aneurysm arising from a nonaneurysmal grossly infected aorta. The affected aortic segment was excised and the intestinal defect was repaired. The aortic stumps were sutured and an axillobifemoral bypass was performed. The patient had an uncomplicated postoperative course.

Primary aortoenteric stula (AEF) between the bowel (usually the duodenum) and the abdominal aorta is a rare and often lethal complication. It is usually caused by erosion of an infrarenal abdominal aortic aneurysm to the third or fourth portion of the duodenum.1 Less commonly it can result from a mycotic aortic aneurysm and even more rarely from infection of a nonaneurysmal aorta, leading to pseudoaneurysm or contained retroperitoneal rupture.2-5 Microbial arteritis is the second most common etiology of infected aneurysms after trauma and is the result of colonization of diseased vessel intima, usually from atherosclerosis, by the microorganism.6 We present the case of a middleaged patient with AEF resulting from septic aortitis and associated pseudoaneurysm. Four more cases have been described in the published data.

CASE REPORT
A 51-year-old male patient was admitted to the emergency department from another hospital with the diagnosis of an infrarenal abdominal aortic aneurysm. He had a history of intermittent low-grade fever during the last 2 months of unknown origin. High fever with chills, episodic abdominal and lumbar pain appeared the week before the admission. Weight loss (7 kg during the last 2 months), anorexia, and weakness were also present. He had not manifested alterations in bowel habits, melena, or vomiting. The patient, a heavy smoking sailor, had no other medical comorbidities. The aneurysm was rst diagnosed by means of an abdominal ultrasound scan. At his admission the patient was septic and hemodynamically stable. Blood tests revealed leucocytosis, elevated inammatory markers, and low albumin level. An abdominal CT scan with i.v. contrast was initially performed revealing a saccular pseudoaneurysm of the infrarenal aorta, whereas the pararenal aorta and the iliac arteries were uninvolved. Splenomegaly was also evident (Figs. 1A, B). Combined parenteral antibiotic treatment was immediately started and parenteral nutrition was also undertaken because of associated malnutrition. Two blood cultures isolated Salmonella spp and Widal test was also positive (B(o) + 1:1600). The patient being stable, an angiogram was performed to assess the lower limb runoff and to evaluate the status of the aorta as 3-D CT-scan

Department of Vascular Surgery, KAT General Hospital, Athens, Greece. Correspondence to: Georgios Skourtis, Department of Vascular Surgery, KAT General Hospital, Nikis 2-4 Kissia, 145 61 Athens, Greece, E-mail: skourtisgeo@in.gr Ann Vasc Surg 2010; 24: 825.e7-825.e11 DOI: 10.1016/j.avsg.2010.02.030 Annals of Vascular Surgery Inc. Published online: May 17, 2010

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Fig. 2. Digital subtraction angiography: lobulated saccular pseudoaneurysm in otherwise normalappearing infrarenal abdominal aorta. through the transverse mesocolon was positioned over the proximal aortic stump, and a closed suction drain was placed in the bed of the excised aortic segment. The duodenal defect was repaired in two layers. After abdominal wall closure, surgical skin preparation and draping were repeated, and an axillobifemoral bypass was performed using a new set of sterilized instruments. An 8mm externally supported PTFE graft was used for this procedure. The time interval between clamping of the abdominal aorta and reperfusion of the lower extremities was 2:35 hours. Cultures of the aortic wall and periaortic tissue had shown growth of Salmonella spp. The patient had an uneventful postoperative course and received intravenous antibiotic regimen (amoxicillin plus clavulanic and ciprooxacin) for 2 weeks. Fever and leucocytosis subsided within 5 days while oral uid administration was started 6 days postoperatively. No major complications occurred, and the patient was discharged after 19 days of hospitalization. Amoxicillin plus clavulanic acid (625 mg/8 hour) P.O. was continued for 3 months. All inammatory markers including the erythrocyte sedimentation rate and C-reactive protein were within normal values 1 month postoperatively. At 6 months, a CT scan of the abdomen was performed without revealing any pathologic ndings except splenomegaly. Ten months after the surgery, the patient is in good clinical condition, regained normal life activities, and continues to be under close surveillance.

Fig. 1. Abdominal CT scan with i.v. contrast: multilobulated pseudoaneurysm of the infrarenal aorta A, adherent to the duodenum with loss of the retroperitoneal tissue planes B. reconstruction was not available. This revealed a lobulated pseudoaneurysm of the aorta near the iliac bifurcation, with normal appearing neighboring vessels (Fig. 2). An urgent surgical management was decided. A few hours before surgery, the patient had an episode of hemodynamic compromise with severe hypotension, intense back pain, and was immediately admitted to the operating room. After induction of anesthesia, a nasogastric tube was introduced and great amount of fresh blood was evacuated. Intraoperatively, a large saccular pseudoaneurysm of the lower abdominal aorta eroding the third portion of the duodenum was found (Figs. 3A, B). The infrarenal aorta was moderately atherosclerotic with marked periadventitial inammation and brosis. Operation included extensive debridement of the aorta and periaortic tissue involved in the inammatory process and suture-ligation of the proximal and distal aortic stump with preservation of the iliac bifurcation. Both ends of the aorta were closed with running monolament interlocking sutures 3-0 polypropylene. An omental pedicle passed

DISCUSSION
Primary AEFs are far less common than secondary stulas.7 The former more frequently result from

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Fig. 3. A, B Separation of the duodenum and the aorta reveals the site of the stula (infrarenal abdominal aorta clamped).

atherosclerotic degenerative abdominal aortic aneurysms that erode to the duodenum. Less common etiologies include aortic dissection, trauma, cancer, gallstones, peptic ulcer, and infection.1,4,8 Microbial arteritis is an infectious process that affects a non-aneurismal artery and develops an aneurysm or arterial rupture with pseudoaneurysm. It is cited as the second most common cause of infected aneurysm. The prevalence in adults is estimated to be 0.06-0.65% and may account for 70-80% of infected aortoiliac aneurysms.9,10 Atherosclerosis of the affected vessel and concurrent bacteremia are involved in the pathogenesis, and the predominant microorganisms are Salmonella species, Staphylococcus species, Escherichia coli, Klebsiella pneumoniae, Mycobacterium tuberculosis, and anaerobic species. In recent reports of infectious aortitis, Salmonella is the offending organism in

18-74% of cases.11,12 The infrarenal aorta is the most common location as it is most frequently affected by atherosclerosis. In addition to our patient there have been seven previous reports describing a primary AEF that resulted from primary septic aortitis with contained retroperitoneal rupture/pseudoaneurysm formation and erosion to the duodenum. The microorganisms involved in these cases included Salmonella species in two, M. tuberculosis in two, Streptococcus viridans in one, and Arizona hinshawii in the oldest report.13-19 Six patients had involvement of the infrarenal abdominal aorta, whereas the pararenal segment was affected in one case17 (Table I). Infected aneurysms are frequently characterized by nonspecic symptoms and signs, and a high index of suspicion is important for early diagnosis. Fever of unknown origin, abdominal and/or back pain, palpable pulsatile abdominal mass, and signs of rupture can be present in various combinations. In cases complicated with aortoenteric erosionstula, most patients initially present with episodic self-limited hemorrhage followed by massive exsanguination. The latter is the rst manifestation in <5% of cases; therefore, time for diagnosis and management is usually available. Diagnosis is difcult and as already mentioned necessitates a high index of suspicion. Upper gastrointestinal endoscopy including the distal duodenum is essential, although it can dislodge fresh thrombus in the stula and induce massive hemorrhage. Radiologic studies include CT scan with i.v. contrast and angiography. CT scan is the most sensitive method for early detection of aortic infection and should be the rst imaging study performed.19 Blood cultures are essential not only for diagnosis but also for appropriate antibiotic treatment. Intraoperative Gram stains and cultures of the aortic wall and periaortic tissues are also important. Patients with infected aortoiliac aneurysms reported in the published data have positive preoperative blood cultures in 65-70%, positive aneurysm wall cultures in 74-92%, and positive Gram stains in 11-50%.20 Treatment of infected aneurysms includes immediate initiation of antibiotics and urgent surgical repair as they are prone to rupture.21,22 During surgery the affected aortic wall should be excised with wide debridement of surrounding infected tissue. When AEF is present the duodenal defect is treated with suture closure in two layers or resection and intestinal anastomosis. Revascularization can be performed by suture-closure of the infrarenal aortic stump combined with an extra-anatomic axillobifemoral bypass or with in-situ aortic reconstruction.7,23,24 The latter can be performed with the use

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Table I. Septic aortitis with pseudoaneurysm and aortoduodenal stula formationdreview of four cases (without preexisting aneurysm)

Characteristics of patients with AEF due to septic aortitis without previous aortic aneurysm

Salmonella enteritidis, group B

Streptococcus viridans Salmonella non-typhimurium

Mycobacterium tuberculosis

Mycobacterium tuberculosis

Salmonella enteritidis

Arizona hinshawii

Etiological agent

of antibiotic-bonded prosthesis, aortic homografts, or supercial femoral veins.12,25,26 The optimal management remains somewhat controversial and is greatly inuenced by the severity of local infection, the presence of gross purulence, and involvement of the suprarenal aorta.27 In our case, the extra-anatomic reconstruction was preferred to avoid the risks of placing a graft in a septic environment as this is reported to be associated with a 23% reoperation rate and even 63% in cases of Gramnegative infection.2,19,23,28 This approach is reported in the published data to have the least postoperative complications and the best survival rate 71% versus 51% compared with in situ revascularization.28 Overall, mortality of patients with primary AEFs is high (reaches up to 70-80%).7 The combination of septic aortitis and AEF may be associated with a more adverse prognosis. Optimal duration of antibiotic treatment is also not well dened with recommendations ranging from 6 weeks to lifelong therapy.27,29,30 The decision should be determined by the severity of the infectious process, the reconstructive procedure selected, the clinical condition of the patient, the course of inammation markers, and the results of radiologic follow-up. Lifelong surveillance is mandatory in these patients.

Alive after 9 months

Alive after 2 years

Alive after 3 years

Alive after 7 years

Death in the operating room Death early postoperatively

Aortic suture ligation + axilobifemoral bypass In situ prosthetic reconstruction In situ prosthetic reconstruction In situ prosthetic reconstruction Aortic suture ligation Axilobifemoral bypass

In situ prosthetic reconstruction

Treatment

Death 19th day postoperatively

Outcome

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Clinical ndings

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22. Wang JH, Liu YC, Yen MY, et al. Mycotic aneurysm due to non-typhi Salmonella: report of 16 cases. Clin Infect Dis 1996;23:743-747. 23. Taylor LM Jr, Deitz DM, McConnell DB, Porter JM. Treatment of infected abdominal aneurysms by extraanatomic bypass, aneurysm excision, and drainage. Am J Surg 1988;155:655-658. 24. Dossa CD, Pipinos I, Shepard AD, et al. Primary aortoenteric stula. Ann Vasc Surg 1994;8:113-120. 25. Trout HH, Kozloff L, Giordano GM. Priority of revascularization in patients with graft enteric stulas, infected arteries or infected arterial prostheses. Ann Surg 1984;199: 669-683. 26. Gupta AK, Bandyk DF, Johnson BL. In situ repair of mycotic abdominal aortic aneurysms with rifampin-bonded gelatin impregnated Dacron grafts: a preliminary case report. J Vasc Surg 1996;24:472-476. 27. Muller BT, Wegener OR, Sandmann W, et al. Mycotic aneurysms of the thoracic and abdominal aorta and iliac arteries: experience with anatomic and extra-anatomic repair in 33 cases. J Vasc Surg 2001;33:106-113. 28. Pasic M, Carrel T, Tonz P, et al. Mycotic aneurysm of the abdominal aorta: extra-anatomic versus in situ reconstruction. Cardiovasc Surg 1993;1:48-52. 29. Chan FY, Crawford ES, Cosseli JS, Sa HJ, Williams TW Jr. In situ prosthetic graft replacement for mycotic aneurysm of the aorta. Ann Thorac Surg 1989;47:193-203. 30. Holher LH, Money SR, Creely B, Bower TC, Kazmier FJ. Direct replacement of thoracoabdominal aortic aneurysms. J Vasc Surg 1993;18:477-485.