Digestive and Liver Disease 35 (2003) 193–196 www.elsevier.com / locate / dld
Brief Clinical Observation
Lower intestinal bleeding due to aorto-enteric fistula C. Maiolo a,b , S. Caprioglio a , G. Cadario a , A. De Lorenzo b , * a
b
Division of Internal Medicine, S. Andrea Hospital, Vercelli, Italy Division of Human Nutrition, Faculty of Medicine and Surgery, University of ‘ Tor Vergata’, Rome, Italy Received 7 February 2002; accepted 6 May 2002
Abstract The case is described of a man who complained of intermittent fever and fatigue. After three digestive endoscopies and computed tomography, a 99m technetium-HM-PAO-labelled white cell scan was usefully employed to establish diagnosis. Anaerobic aortic Graft infection and anaemia due to lower intermittent occult intestinal bleeding were found. The intestinal bleeding was caused by secondary aorto-jejunal fistula. This condition is rare, but should be suspected whenever a patient with aortic prosthesis presents with occult digestive bleeding and unexplained fever. 2003 Editrice Gastroenerologica Italiana S.r.l. Published by Elsevier Science Ireland Ltd. All rights reserved. Keywords: Anaemia; Aorto-jejunal fistula; Intestinal bleeding; Vascular graft infection
1. Introduction Prosthetic graft infections are uncommon complications of aortic bypass (incidence between 1 and 6%) and may result in serious sequelae such as limb loss. The sequelae of these infections may even be lethal [1]. Conditions associated with prosthetic vascular infection include pseudoaneurysm, hydronephrosis, osteomyelitis, and vascular enteric fistulas. Specifically, aorto-enteric anastomotic fistula represent a life-threatening complication of abdominal aortic surgery [2]. Although the incidence of these complications is reported to be rare, the difficulty in diagnosis and management has led to high mortality rates and extensive morbidity [3]. It has been demonstrated that grafts implanted in the inguinal area have a higher rate of infection than those that lie entirely within the abdomen and that infection of the intra-abdominal extremities of vascular grafts is most frequently associated with prostheto-enteric fistula [4]. The case is described here of prosthetic infection complicated by aorto-enteric fistula with an uncommon presentation, namely, anaemia and fever. To our knowl*Corresponding author. Tel.: 139-06-7259-6415; fax: 139-06-72596407. E-mail address:
[email protected] (A. De Lorenzo).
edge, this is the first case report of a prosthesis infection presenting as an early bacteraemic spread of Clostridium glycolicum and anaemia caused by secondary aorto-jejunal fistula.
2. Case report A 67-year-old man was referred to the Internal Medicine Division on account of fatigue and intermittent fever. The patient, with a past history of hypertension and myocardial infarction, had Claudicatio Intermittens due to Leriche’s Syndrome in 1994. In that same year, he, therefore, underwent aortic-bifemoral bypass. In December 2000, he complained of fatigue, anorexia, mild weight loss, and intermittent fever up to 38.5 8C. Two months later, in February 2001, he was admitted to hospital on account of anaemia (haemoglobin: 7.3 g / dl, MCV: 86 fl, MCHC: 31.8 g / dl). The patient was thus given four units of packed red-blood cells. Haemoglobin rose initially, but later decreased. On admission, he was pale and asthenic, but afebrile. Systolic murmurs were heard in the femoral bilateral regions. Rectal exploration revealed normal stool. His home care treatment consisted of calcium-antagonist drugs and low-dose aspirin. Laboratory examinations were as follows: white blood cells (WBCs) 6.25310 9 / l, serum
1590-8658 / 03 / $30 2003 Editrice Gastroenerologica Italiana S.r.l. Published by Elsevier Science Ireland Ltd. All rights reserved. doi:10.1016 / S1590-8658(03)00028-8
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iron 16 ng / dl, transferrin 2.02 g / l, ferritin 23 ng / ml, bilirubin 0.3 mg / dl, lactate dehydrogenase 117 U / l, erythrocyte sedimentation rate 28 mm / h, C-reactive protein 0.30 mg / dl. Out of 3 samples of faecal occult blood, 2 resulted positive. Evaluations of circulating IgA and IgG antigliadin, and antiendomysial antibodies were negative. High charge of Clostridium glycolicum was isolated from one out of six haemocultures. Gastric-duodenal-endoscopy did not reveal any mucosal lesions nor any source of active bleeding. Biopsies of the duodenal mucosa were collected but no evidence of coeliac disease was found. Enteroscopy with a paediatric size colonoscope, permitting inspection of the entire duodenum
and of part of the jejunum, did not reveal any bleeding sites. Furthermore, colonoscopy did not show lowlying lesions such as bleeding haemorrhoids, polyps, colon cancer, or colon haemorrhagic angiodysplasia. A contrastenhanced computer tomography (CT) scan of the abdomen showed an infra-renal abdominal aortic aneurysm and almost total occlusion of the left aortic-femoral allo-graft by-pass. In addition, dense tissue between a peri-prosthetic area on the left side and a tract of the small intestine was observed. Later, in May, 2001, an isotopic study with labelled 99m Tc-HM-PAO WBC scan (Fig. 1) was performed. This test clearly showed the presence of labelled WBC in the left
Fig. 1. In anterior (left side) and posterior (right side) 99m Tc-HM-PAO labelled WBC scan shows progressive (after 3 and 20 h) and extensive uptake in left groin (graft infection).
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graft prosthesis and confirmed the suspected aorta-iliac graft infection. After controlling the infection, the patient was sent to the vascular surgeon for the replacement of the left aortic-femoral prosthesis. At surgery, a large left inguinal graft abscess was discovered, a sample of which was sent for micro-biological analysis; the left aortic graft was excised and the lower left limb revascularized with an extra-anatomical right axillofemoral bypass. Furthermore, during surgery, an enteric (distal jejunum)–left aortic (para-prosthesis) fistula was discovered. The next steps in the surgical treatment were perforated jejunum tract resection and latero-lateral jejuno-jejunostomy, from which the patient recovered satisfactorily, without complications. Culture of the graft abscess, sampled at surgery, isolated the Gram-negative enteric bacillus Escherichia coli. Finally, one month later, after specific anti-microbial treatment, no further episodes of fever or anaemia have occurred.
3. Discussion In our patient, the aortic prosthetic infection was complicated by aorto-jejunal fistula presenting as intermittent fever and anaemia. One of the several episodes of fever was found to be associated with the bacteraemic spread of Clostridium glycolicum. Most graft infections are caused by skin commensals such as Staphylococcus aereus and Staphylococcus epidermidis. Anaerobic prosthetic infection is an unusual event and aortic graft infection due to Clostridium has rarely been reported [1,5]. Despite the many specific imaging techniques available, the diagnosis of graft infections remains difficult, particularly as far as intra-abdominal grafts are concerned, due to the non-specific clinical and imaging findings [6]. The 99m Tc labelled WBC scan is an important complementary test to CT in ambiguous cases and may be more sensitive in detecting graft infections. Most graft infections occur as a result of the direct contamination of the prosthesis at the time of surgery, the usual source of bacteria being the patient’s skin [7]. However, Hannon et al. demonstrated that, in a series of cases of aortic graft infection from one unit, more than half of the grafts cultured grew enteric bacteria [8]. This case extends the clinical spectrum of Clostridium infections by documenting a prolonged interval between the prosthesis implantation and its subsequent infection (six years). Anaemia was the other manifestation of the patient’s disease. This was found to be derived from intermittent occult intestinal bleeding. The patient was having recurrent acute bleeding episodes typical of recurrent sentinel bleeds from a fistula. Aorto-enteric fistula, specifically with jejunal involvement, was suspected. Aorto-enteric fistula should be considered a subset of aortic graft infection [6]. On the other hand, secondary aorto-jejunal fistulas are the rarest aorto-enteric fistulas, primary aorto-duodenal fistulas
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being the most frequent. Primary fistulas occur after aneurysms of the native aorta, while secondary fistulas rarely occur after placement of an aortic prosthesis [9]. In most patients with aorto-enteric fistulas, clinical manifestations include abdominal pain or mass, sometimes fever, maelena, or haematemesis. No such symptoms were observed in our patient and endoscopic findings resulted negative for bleeding. Therefore, anaemia originating from jejunal bleeding was the presenting sign of the aorticenteric fistula, acquired through prosthetic infection. In addition, the chronic infection was part of the cause of anaemia. In a recent study, aorto-duodenal fistula was described complicating an infected aortic graft, the diagnosis of which was reached by means of leukocyte scintigraphy [10]. Successful management of aorto-enteric fistulas requires a high index of suspicion, for early diagnosis. Although secondary aorto-jejunal fistulas are rare, their presence should be suspected whenever a patient with aortic prosthesis has digestive occult bleeding and unexplained fever.
Conflict of interest statement None declared.
List of abbreviations CT, computed tomography; WBCs, white blood cells.
Acknowledgements Authors thank Dr. M. De Allegri for review of manuscript, and Dr. C. Pollo, Radiology Division, Dr. R. Cantone, and Dr. V. Semeraro, Internal Medicine Division of S. Andrea Hospital, Vericelli, for collaboration.
References [1] Holland FW, Darling RC, Chang BB, Shah DM, Leather RP. Clostridial aortic graft infection. Ann Vasc Surg 1994;8:387–9. [2] Antinori CH, Andrew CT, Santaspirt JS, Villanueva DT, Kuchier JA, DeLeon ML, et al. The many faces of aortoenteric fistulas. Am Surg 1996;62:344–9. [3] Gutowski P, Szumilowicz G, Cnotliwy M. Evaluation of various diagnostic methods in aortic-iliac graft infection. Wiad Lek 1997;50:27–31. [4] Parola P, Alimi Y, Juhan C, Brouqui P. Infections of vascular prostheses of the abdominal aorta. Diagnostic and therapeutic problems. J Mal Vasc 1999;24:194–201. [5] Feldman RI, Kallich M, Weinstein MP. Bacteremia due to Clos-
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tridium difficile: case report and review of extraintestinal C. difficile infection. Clin Infect Dis 1995;20:1560–2. [6] Orton DF, LeVeen RF, Saigh LA, Culp WC, Fidlen JL, Lch TJ, et al. Aortic prosthetic graft infections: radiologic manifestations and implications for management. Radiographics 2000;20:977–93. [7] Goldstone J, Bowersox JC. Infected prosthesis arterial graphs. In: Haimovici H, Ascer E, Holier LH, Strandness DE, Towne JB, editors, Haimovici’s Vascular Surgery, Blackwell Science, 1996, pp. 727–39.
[8] Hannon RJ, Wolfe JHN, Mansfield AO. Aortic prosthetic infection: 50 patients treated by radical or local surgery. Br J Surg 1996;83:654–8. ˆ F, Conri C. Secondary [9] Constans J, Midy D, Baste JC, Demortiere aorto-duodenal fistulas: report of 7 cases. Rev Med Int 1999;20:121–7. [10] Ganatra RH, Haniffa MA, Hawthorne AB, Rees JI. Aortoenteric fistula complicating an infected aortic graft: diagnosis by leukocyte scintigraphy. Clin Nucl Med 2001;26:800–1.
