Hrev_master [page 44] [Emergency Care Journal 2016; 12:5845] The cause of intestinal bleeding cannot be missed: the case of primary aortoenteric fistula Bahjat Barakat,1 Lucia Calculli,2 Raffaele Pezzilli3 1Department of Emergency; 2Department of Diagnostic Medicine and Prevention; 3Department of Digestive System, Sant’Orsola-Malpighi Hospital, University of Bologna, Bologna, Italy Abstract Primary aortoenteric fistula (PAEF) is a rare condition of intestinal hemorrhage. We herein report a case of this condition initially suspect- ed by ultrasonographic evaluation and subse- quently confirmed by computed tomography. PAEF should be considered a possible cause in patients with gastrointestinal bleeding of uncertain etiology and especially in patients with known aortic aneurysms. The rapid recog- nition of this condition may save the life of these patients because in untreated subjects the mortality is 100%, frequently due to hypov- olemic shock. Introduction Gastrointestinal hemorrhage represents a challenge in Emergency Room. Upper gastro- intestinal bleeding is a common medical emer- gency ranging from 50-150 cases per 100,000 inhabitants.1 Most of these bleeding are non- varicose (esophagitis, Mallory-Weiss syn- drome, peptic ulcer, gastric neoplasia, angiodysplasia); primary aorto-enteric fistula is a rare entity but has a high mortality rate and requires early diagnosis for the proper treatment. The accurate clinical history may select those patients having hemorrhage asso- ciated to other pathological conditions such as variceal bleeding whereas sometimes the hemorrhage may be due to rare cause. We report a case of massive intestinal bleeding due to a rare condition such as primary aorto- enteric fistulas (PAEF). Case Report A 91-year-old man was admitted to the Emergency Room for intestinal bleeding, tachycardia and hypotension (high cardiac rate 120 b.p.m. and arterial blood pressure 100/60 mmHg). His past medical history was unremarkable except for an operation for a pharynx tumor and for the presence of benign prostate hypertrophy. Physical examination revealed a pulsatile mass of the abdomen and the presence of massive rectal bleeding. Patient underwent to fluid resuscitation and blood analyses revealed normal hemoglobin. An ultrasonography of abdomen revealed an abdominal aortic aneurysm of about 10 cm of diameter partially thrombosed (Figure 1A and B); a dilation of small bowel loops, and the presence of fluid-fluid levels (Figure 2). Echo- color-Doppler confirmed the aneurysm with thrombosis (Figure 1C and D). One hour after admission, the patient general condition start- ed to deteriorate and he became hemodinami- cally unstable, the hemoglobin dropped to 8 mg/dL and 4 units of blood transfusion was administered. After patient stabilization a con- trast enhanced-CT scan was carried out. This examination confirmed the presence of a sub- renal aortic aneurysm measuring 10 x 9 cm with a filiform aortic-enteric fistulous tract and leakage of contrast material in the stom- ach and ileum (Figures 2 and 3). The patient was immediately transferred to the Emergency Surgical Department where the patient was operated. A prosthetic Goretex tube graft was inserted in the aorta and the fis- tula, and the intestine was repaired by direct suturing. The patient had an uneventful recovery and was discharged home after seven days in sta- ble condition. Discussion Primary aortoenteric fistula (PAEF) should be considered a possible cause of gastro-intesti- nal bleeding of uncertain etiology and espe- cially in patients with known abdominal aortic aneurysm.2 PAEF is a rare entity and the reported inci- dence of PAEF is 0.007 per million3 and it is associated with a high mortality. Its occur- rence is usually due to erosion of an abdominal aortic aneurysm into the intestinal tract. The most common sites for the fistula are the third and fourth portions of duodenum2 and it has also been reported the presence of fistula to the esophagus, stomach, small bowel, colon.1 A classical triad of this condition, i.e. gas- trointestinal hemorrhage, abdominal mass and abdominal or back pain is uncommon.4-7 As in our case, the typical bleeding pattern associat- ed with PAEF is usually intermittent, starting with a brief herald bleeding followed by major gastrointestinal hemorrhage having often a fatal outcome.8 Even if the preoperative exam- inations are often not helpful and can lead to delayed diagnosis and surgery, we suspected the presence of PAEF by a simple imaging technique such as transabdominal ultrasonog- raphy associated with echo-color-Doppler and the final diagnosis was made by contrast- enhanced computed tomography that showed the presence of abdominal aneurysm, the link- age between the aneurysm and the duodenum and the presence of blood in the intestinal lumen. In our patient, as usually happens in these subjects, there were risk factors for the presence of atherosclerosis and an upper gas- trointestinal bleeding in the absence of an evi- dent source. Definitive treatment includes pri- mary duodenal repair and insertion surgically or radiologically of an aortic stent9-11 or aortic aneurismal resection.12 Conclusions PAEF should be considered a possible cause in patients with gastrointestinal bleeding of uncertain etiology and especially in patients with known aortic aneurysms. The rapid recog- nition of this condition, that should be consid- ered a true abdominal vascular catastrophe,13 saves the life of these patients because in untreated subjects the mortality is 100%, fre- quently due hypovolemic shock.11 Emergency Care Journal 2016; volume 12:5845 Correspondence: Raffaele Pezzilli, Department of Digestive System, Sant’Orsola-Malpighi Hospital, Bologna, Italy. Tel: +39.051.6364148 - Fax: +39.051.6364148. E-mail: raffaele.pezzilli@aosp.bo.it Key words: Primary aortic-enteric fistula; Intestinal bleeding; Ultrasonography; Color- Doppler; Computed tomography. Conflict of interest: the authors decalre no poten- tial conflict of interest. Received for publication: 25 February 2016. Revision received: 26 May 2016. Accepted for publication: 26 May 2016. This work is licensed under a Creative Commons Attribution 4.0 License (by-nc 4.0). ©Copyright B. Barakat et al., 2016 Licensee PAGEPress, Italy Emergency Care Journal 2016; 12:5845 doi:10.4081/ecj.2016.5845 Non co mmerc ial us e o nly [Emergency Care Journal 2016; 12:5845] [page 45] References 1. Romera Barba E, Sánchez Pérez A, Bertelli Puche J, et al. Primary aorto-duodenal fis- tula: a rare but potentially fatal cause of gastrointestinal bleeding. Cir Espan 2015;93:121-3. 2. Alzobydi AH, Guraya SS. Primary aortoduo- denal fistula: a case report. World J Gastroenterol 2013;19:415-7. 3. Parikh K, Ali MA, Wong RC. Unusual caus- es of upper gastrointestinal bleeding. Gastrointest Endosc 2015;25:583-605. 4. Saers SJ, Scheltinga MR. Primary aortoen- teric fistula. Brit J Surg 2005;92:143-52. 5. Delgado J, Jotkowitz AB, Delgado B, et al. Primary aortoduodenal fistula: pitfalls and success in the endoscopic diagnosis. Eur J Intern Med 2005;16:363-5. 6. Ihama Y, Miyazaki T, Fuke C, et al. An autopsy case of a primary aortoenteric fis- tula: a pitfall of the endoscopic diagnosis. World J Gastroenterol 2008;14:4701-4. 7. Lee CW, Chung SW, Song S, et al. Double primary aortoenteric fistulae: a case report of two simultaneous primary aortoenteric fistulae in one patient. Korean J Thorac Cardiovasc Surg 2012;45:330-3. 8. Nohr M, Juul-Jensen KE, Balslev IB, Jelnes R. Primary aorto-enteric fistula: a practica- ble curable condition? Pathogenetic and clinical aspects. Int Angiol 1990;9:278-81. 9. Cendan JC, Thomas JB 4th, Seeger JM. Twenty-one cases of aortoenteric fistula: lessons for the general surgeon. Am Surg 2004;70:583-7. 10. Shapiro M, Addis MD, Ellozy SH, et al. Successful endovascular treatment of bleeding aortoenteric fistula: a case report. Ann Vasc Surg 2006;20:817-9. 11. Khang NC, Zainal AA. Endovascular stent graft repair of aorto-iliac pseudoa- neurysms - Hospital Kuala Lumpur experi- ence. Med J Malaysia 2016;71:17-22. 12. Rodrigues dos Santos C, Casaca R, Mendes de Almeida JC, Mendes-Pedro L. Enteric repair in aortoduodenal fistulas: a forgotten but often lethal player. Ann Vasc Surg 2014;28:756-62. 13. Singh M, Koyfman A, Martinez JP. Abdominal vascular catastrophes. Emerg Med Clin N Am 2016;34:327-39. Case Report Figure 1. Typical ultrasonographic appearance of abdominal aortic aneurysm: A) longi- tudinal scan; B) axial scan with clear evidence of the parietal thrombosis delimiting the true lumen; C) and D) echo-color-Doppler confirming the aneurysm with thrombosis. Figure 2. Abdominal ultrasonography scan of the central abdomen showing dilated fluid-filled small bowel loops with thick- ened wall. Figure 3. A) Abdominal contrast enhanced computer tomography showing aortic aneurysm of the abdominal aorta with a penetrating right anterolateral ulcer (red arrow); B) the aneurysm causes compressive effect and disappearance of a fat cleavage plane of the duodenum; the duodenum is hyperdense in its inferior-external wall in relation to the hemorrhage. Non co mmerc ial us e o nly