- Email: [email protected]
Primary Aortoduodenal Fistula in a Patient With Pararenal Abdominal Aortic Aneurysm
Primary Aortoduodenal Fistula in a Patient With Pararenal Abdominal Aortic Aneurysm Beatriz Genoves-Gasco, Alvaro Torres-Blanco, Angel Plaza-Martı´nez, David Olmos-Sanchez, Francisco Gomez-Palones, and Eduardo Ortiz-Monzon, Valencia, Espa~ na
Primary aortoenteric fistula is a rare and extremely serious condition. In most cases, it is caused by an abdominal aortic aneurysm presenting with symptoms of gastrointestinal bleeding. Diagnosis is difficult owing to its rarity and the fact that diagnostic tests are not definitive in many cases. Surgery is performed urgently in most cases and is associated with high mortality. We report a case of a 65-year-old man presenting with symptoms of abdominal pain and massive rectal hemorrhage. Computed tomography revealed a pararenal abdominal aortic aneurysm and suspected aortoenteric fistula. The patient underwent an emergency surgery, confirming the suspected diagnosis. The surgery performed was the traditionally recommended extraanatomical bypass with aortic ligation and repair of the intestinal defect. We describe the clinical condition and provide an up-to-date overview of diagnosis and treatment by reviewing the literature. We believe the therapeutic decision should be personalized by assessing the anatomy of the aneurysm, the patient’s clinical status, the degree of local contamination, and the surgeon’s experience with each of the techniques.
Aortoenteric fistula (AEF)ecommunication between the aorta and the intestinal lumeneis a very rare and extremely serious condition, which requires emergency treatment in most cases. Despite the different surgical treatment strategies, it still has a very high morbidity and mortality. The AEF may be primary, especially associated with aortic aneurysms, or secondary, more frequent and associated with previous aortic surgery using prosthetic graft. Clinical manifestation is usually gastrointestinal bleeding, sometimes associated with signs of sepsis. Classical treatment with conventional surgery is still in effect, and the aortic ligature and extraanatomical bypass is the most recommended option, although this presents the disadvantage of a high morbidity and mortality associated with it.1e5
Servicio de Angiologı´a y Cirugı´a Vascular, Hospital Universitario Dr. Peset, Valencia, Espa~ na. Correspondence to: Beatriz Genoves-Gasco, MD, Servicio de Angiologı´a y Cirugı´a Vascular, Hospital Universitario Dr. Peset. Avda. Gaspar Aguilar, 90. 46017 Valencia, Espa~ na; E-mail: [email protected] Ann Vasc Surg 2012; 26: 730.e1e730.e5 DOI: 10.1016/j.avsg.2011.11.030 Ó Annals of Vascular Surgery Inc. Published online: April 13, 2012
Conversely, endovascular treatment is a promising option, especially in emergency situations and in patients at high surgical risk owing to its minimally invasive nature.6 It is also especially attractive in case of active or recent hemorrhage and hemodynamic instability.7 Excellent results have been reported in the short-term in many cases,8e10 although in the long-term, the problem of prosthetic graft infection does not seem to be resolved.6,7 In addition, it is not possible to perform endovascular treatment in all patients owing to the anatomical requirements needed for a correct fixation and proper sealing of the endograft.
CLINICAL CASE A 65-year-old man with a personal history of dyslipidemia, chronic obstructive pulmonary disease, and inguinal herniorrhaphy arrived at the emergency department in an ambulance after an episode of abdominal pain and massive rectal bleeding with loss of consciousness. During his stay in the emergency department, he presented with sustained hypotension and an episode of hematemesis. Physical examination revealed a pulsatile abdominal mass, objectifying the existence of an abdominal 730.e1
730.e2 Case reports
aortic aneurysm (AAA) on abdominal ultrasonography. Computed tomography (CT) with intravenous contrast showed a fusiform pararenal AAA with a maximum diameter of 92 mm, which included the left renal artery and extended up to the iliac bifurcation. We also observed a duodenal loop in close contact with its anterior wall, with possible solution of continuity (Fig. 1). With the suspected diagnosis of AEF, the patient underwent an emergency open surgery. We performed an initial axillobifemoral bypass from the right axillary artery using a prosthetic expanded polytetrafluorethylene graft. Subsequently, we accessed the retroperitoneum by means of median laparotomy. First, we proceeded to dissect the aortic neck and both primitive iliac arteries. During the process of detachment of the duodenal loop from the anterior wall of the aneurysm, we discovered the AEF in the third duodenal portion (Fig. 2). We sutured the duodenal wall aneurysm defect and performed the resection of the aneurysm after proximal aortic clamping between the renal arteries. A proximal infrarenal suture of the aneurysm was then performed (double continuous polypropylene suture) and a portion of omentum was wrapped around the closure site. During surgery, the patient remained hemodynamically stable. After surgery, in the intensive care unit, the patient remained sedated, adapted to mechanical ventilation, and started to present hemodynamic instability, requiring vasoactive drugs. Clinical course from the outset showed a slow healing pattern, with gradual multiorgan dysfunction associated with renal, respiratory, and hemodynamic failure. Owing to the unfavorable clinical course and findings on CT of fluid in the peritoneal cavity, dehiscence of the abdominal suture was suspected, and an exploratory laparotomy was performed at 4 days after the admission. Serous fluid was drained, with no macroscopic appearance of an infection, and verifying that intestinal suture presented a good aspect without dehiscence. Subsequently, during his stay in the intensive care unit, the patient developed episodes of nosocomial infection, with Pseudomonas aeruginosa and Acinetobacter baumannii present in the bronchial secretions. Empirical antibiotic therapy was administered in the beginning, and later the therapy was chosen according to antibiogram. On the 12th day, CT again revealed the existence of abdominal fluid, and hence a percutaneous drainage was positioned. After a period with no significant clinical or hemodynamic alterations, the patient presented with progressive worsening on day 27 after the
Annals of Vascular Surgery
Fig. 1. Computed tomography with intravenous contrast. Duodenal loop adhered to the anterior wall of the abdominal aortic aneurysm (AAA).
Fig. 2. Intraoperative photograph of the AAA and aortoduodenal fistula. (1) Duodenal wall defect. (2) Aortic wall defect.
admission. Nonrecoverable cardiac arrest occurred on the 32nd day in a state of distributive shock.
DISCUSSION Primary AEF is a rare condition typically characterized by the clinical triad of gastrointestinal bleeding, abdominal pain, and pulsatile mass, although the complete triad is only present in a minority of patients.1 In most cases, they are associated with aneurysmatic aortas and present as repeated gastrointestinal bleeding. The time interval between the
Vol. 26, No. 5, July 2012
first bleeding and massive bleeding may range from hours to even months. The incidence reported in the literature is between 0.04% and 0.07%.11,12 In the case of primary AEF, there is a communication between the gastrointestinal tract and the aorta, without a history of surgery (we otherwise define this as secondary). Approximately 80% of cases are caused by an AAA, and atherosclerosis is the most common etiology. Other causes of AEF are mycotic or traumatic aneurysms, radiation, neoplasias, peptic ulcers, inflammatory processes such as appendicitis or diverticulitis, cystic medial necrosis, or ingestion of foreign bodies. Microorganisms more commonly responsible for mycotic aneurysms are Salmonella or Klebsiella, although Mycobacterium tuberculosis, Treponema pallidum, fungi, Staphylococcus, and Streptococcus, among others, have also been found.13,14 Although primary AEF can affect any part of the gastrointestinal tract, the duodenum is involved in 82% of cases, especially the third and fourth portion.15 Mortality rates are extremely high, close to 100% in untreated cases or those with active bleeding, and 30% to 40% in cases treated by surgery.11,13,16 Diagnosis is especially difficult because it is a very rare entity; classical clinical symptoms are present in a minority of patients (11%),17 and diagnostic tests are not definitive in a significant percentage of cases. In cases of gastrointestinal bleeding, the first recommended diagnostic test is endoscopy; however, it should be performed in hemodynamically stable patients.13,18,19 This pathology should be suspected in the presence of active bleeding, an ulcer, an erosion with an adherent clot, or a pulsatile extrinsic mass. Endoscopy is also very useful to exclude other causes of bleeding such as gastroduodenal ulcers or esophageal varices and offers therapeutic possibilities,13 although it is only suggestive of AEF in 25% of patients13,18 and is not available urgently at many hospitals. With arteriography, we only find extravasation of contrast within the gastrointestinal tract in 26% of cases; however, this defines arterial anatomy and is useful for aortic reconstruction planning.2,5 Currently, with the progress of CT in terms of speed, quality of the image, and resolution and owing to its greater availability and it being a less invasive diagnostic test, it has been proposed as the test of choice and is being used initially to confirm the suspicion of this entity.13,14 Gastrointestinal bleeding in the presence of a known AAA suggests an AEF, and an emergency CT with intravenous contrast is indicated,11 the diagnostic success rate of which is 60%. Presence of air in the aorta or
Case reports 730.e3
contrast in gastrointestinal tract is a highly suggestive image. For an early diagnosis, a high rate of suspicion is essential, especially in patients with AAA who present with gastrointestinal hemorrhage. In the absence of knowledge of the cause of massive hemorrhage, it is recommended to perform an emergency exploratory laparotomy.18 Monitoring blood pressure with maintenance of systolic pressure between 60 and 100 mm Hg may reduce the risk of recurrent bleeding during the period before surgical repair.11 The three goals of surgery are to control the hemorrhage, to control the infection, and to keep distal perfusion.12 As surgical treatment, extraanatomic bypass is recommended to avoid contamination of the prosthesis and to repair the intestinal defect.13 The in situ graft using a polytetrafluorethylene or polyester prosthesis is suitable in unstable patients and in cases where the level of contamination is low. Another option is reconstruction with cryopreserved grafts, but it has disadvantages such as the possibility of rupture or aneurysm formation, a higher cost,20,21 and difficult availability, especially in emergency situations. A portion of the omentum should be interposed between the vascular anastomosis and the adjacent bowel.11,13 In contrast, endovascular surgery can enable emergency exclusion in an unstable patient using a minimally invasive approach and, together with antibiotics, may be a solution even during long periods. There are reported cases of AEF successfully treated with endovascular exclusion,22e25 and some authors suggest it could be the treatment of choice in cases without signs of sepsis.6,12,24,25 Even then, it is not still considered a definitive treatment, although it may serve as a bridge to the generally recommended procedure ‘‘perform secondary and deferred open surgery.’’ This is because with endovascular treatment, the risk of fulminant sepsis and recurrent bleeding remains owing to a persistent source of untreated infection.7,11,26e28 Furthermore, this needs more strict anatomic requirements than open surgery, and the endograft is not always available on an urgent basis. In our case, the absence of a proximal neck of the aneurysm, which would enable proper proximal fixation of the endograft and therefore performing correct endovascular exclusion, enabled us to rule out this option from the outset (Fig. 3). The absence of the aneurysm’s proximal neck also significantly hinders with performing proximal aortoprosthetic anastomosis and encouraged not performing in situ reconstruction, which is better tolerated hemodynamically and recommended by some authors in
730.e4 Case reports
Annals of Vascular Surgery
diagnosis of AEF should be ruled out first in patients with significant gastrointestinal bleeding and known aortic aneurysm. The discussion among the different surgical options is still open. The decision to be made between these alternatives, including endovascular treatments, should be personalized by assessing a series of variables: the advantages and disadvantages of each one of the techniques, the anatomy of the aneurysm, the patient’s clinical status, the degree of infection or local contamination, and the surgeon’s personal experience. REFERENCES
Fig. 3. Computed tomography with intravenous contrast. Absence of the infrarenal neck of the AAA.
cases of not very significant contamination. Therefore, given the anatomical characteristics, we opted for the classical surgical treatment, that is, aortic ligature and extra-anatomical bypass. Although this is the most recommended treatment and the patient was kept stable at all times during surgery, he began to become hemodynamically unstable during the immediate postoperative period. It is precisely this problemdthe hemodynamic repercussiondthat is the main disadvantage of this surgical option. With regard to the intestinal repair, we opted for primary closure given the limited defect, although local resection may be required in the cases of major defects. The result depends on the delay in diagnosis, the patient’s general condition, the degree of contamination, and the location of the complication in the aorta.17 Furthermore, antibiotic therapy should be initiated since the suspicion of AEF, first empirical against the microorganisms described earlier in the text and subsequently specific according to the result of the culture tests. This has to be maintained for 7 to 10 days if culture results are negative and 4 to 6 weeks if they are positive.13,14 A longer duration of the therapy or even antibiotic treatment for a lifetime is recommended in cases of endovascular exclusion.26 In summary, early diagnosis and surgery are vitally important for the patient’s survival. The
1. Saers SJ, Scheltinga MR. Primary aortoenteric fistula. Br J Surg 2005;92:143e52. 2. Bergqvist D. Arterioenteric fistula. Review of a vascular emergency. Acta Chir Scand 1987;153:81e6. 3. Montgomery RS, Wilson SE. The surgical management of aortoenteric fistulas. Surg Clin North Am 1996;76:1147e57. 4. Busuttil SJ, Goldstone J. Diagnosis and management of aortoenteric fistulas. Semin Vasc Surg 2001;14:302e11. 5. Goshtasby P, Henriksen D, Lynd C, et al. Recurrent aortoenteric fistula: case report and review. Curr Surg 2005;62:638e43. 6. Burks JA Jr, Faries PL, Gravereaux EC, et al. Endovascular repair of bleeding aortoenteric fistulas: a 5-year experience. J Vasc Surg 2001;34:1055e9. 7. Chuter TA, Lukaszewicz GC, Reilly LM, et al. Endovascular repair of a presumed aortoenteric fistula: late failure due to recurrent infection. J Endovasc Ther 2000;7:240e4. 8. Allen RC, Sebastian MG. The role of endovascular techniques in aortoesophageal fistula repair. J Endovasc Ther 2001;8:602e3. 9. Semba CP, Sakai T, Slonim SM, et al. Mycotic aneurysms of the thoracic aorta: repair with use of endovascular stentgrafts. J Vasc Interv Radiol 1998;9:33e40. 10. Leobon B, Roux D, Mugniot A, et al. Endovascular treatment of thoracic aortic fistulas. Ann Thorac Surg 2002;74: 247e9. 11. Chaikof EL, Brewster DC, Dalman RL, et al. The care of patients with an abdominal aortic aneurysm: The Society for Vascular Surgery practice guidelines. J Vasc Surg 2009;50:S2e49. 12. Baril DT, Carroccio A, Ellozy SH, et al. Evolving strategies for the treatment of aortoenteric fistulas. J Vasc Surg 2006;44: 250e7. 13. Lemos DW, Raffetto JD, Moore TC, Menzoian JO. Primary aortoduodenal fistula: a case report and review of the literature. J Vasc Surg 2003;37:686e9. 14. San Norberto-Garcı´a EM, Carrera-Dı´az S, Cenizo-Revuelta N, et al. Fı´stula aortodueodenal primaria: descripci on de un caso y revisi on de la bibliografı´a. Angiologı´a 2007;59:445e9. 15. Farber A, Grigoryants V, Palac DM, et al. Primary aortoduodenal fistula in a patient with a history of intravesical therapy for bladder cancer whith bacillus Calmette-Guerin: review of primary aortoduodenal fistula without abdominal aortic aneurysm. J Vasc Surg 2001;33:868e73. 16. Sweeney MS, Gadacz TR. Primary aortoduodenal fistula: manifestations, diagnosis and treatment. Surgery 1984;96: 492e7. 17. Roche-Nagle G, O’Donnell DH, Brophy DP, Barry MC. Primary aortoenteric fistula. Am J Surg 2008;195:506e7.
Vol. 26, No. 5, July 2012
18. Song Y, Liu Q, Shen H, et al. Diagnosis and management of primary aortoenteric fistulas-experience learned from eighteen patients. Surgery 2008;143:43e50. 19. Barkun A, Fallone CA, Chiba N, et al. A Canadian clinical practice algorithm for the management of patients with nonvariceal upper gastrointestinal bleeding. Can J Gastroenterol 2004;18:605e9. 20. Bisdas T, Wilhelmi M, Haverich A, Teebken OE. Cryopreserved arterial homografts vs silver-coated Dacron grafts for abdominal aortic infections with intraoperative evidence of microorganisms. J Vasc Surg 2011;53:1274e81. 21. Bustamante M, G omez-Dermit V, Garcı´a I, et al. Endoluminal repair of a pseudoaneurysm in a patient with cryopreserved arterial allograft of the iliac vessel. Ann Vasc Surg 2009;23:410.e17e20. 22. Gonz alez-Fajardo JA, Gutierrez V, Martı´n-Pedrosa M, et al. Endovascular repair in the presence of aortic infection. Ann Vasc Surg 2005;19:95e8.
Case reports 730.e5
23. D’Ancona G, Dagenais F, Bauset R. Endoluminal stenting of the aorta as treatment of aortoesophageal fistula due to primary aortic disease. Tex Heart Inst J 2002;29:216e7. 24. Deshpande A, Lovelock M, Mossop P, et al. Endovascular repair of an aortoenteric fistula in a high-risk patient. J Endovasc Surg 1999;6:379e84. 25. Eskandari MK, Makaroun MS, Abu-Elmagd KM, Billiar TR. Endovascular repair of an aortoduodenal fistula. J Endovasc Ther 2000;7:328e32. 26. Antoniou GA, Koutsias S, Antoniou SA, et al. Outcome after endovascular stent graft repair of aortoenteric fistula: a systematic review. J Vasc Surg 2009;49:782e9. 27. Danneels MI, Verhagen HJ, Teijink JA, et al. Endovascular repair for aortoenteric fistula: a bridge to far or a bridge to surgery? Eur J Vasc Endovasc Surg 2006;32:27e33. 28. Schlensak C, Doenst T, Slonim SM, et al. Palliative treatment of a secondary aoroduodenal fistula by stent-graft placement. Thorac Cardiovasc Surg 2000;48:41e2.
Primary aortoenteric fistula is a rare and extremely serious condition. In most cases, it is caused by an abdominal aortic aneurysm presenting with symptoms of gastrointestinal bleeding. Diagnosis is difficult owing to its rarity and the fact that diagnostic tests are not definitive in many cases. Surgery is performed urgently in most cases and is associated with high mortality. We report a case of a 65-year-old man presenting with symptoms of abdominal pain and massive rectal hemorrhage. Computed tomography revealed a pararenal abdominal aortic aneurysm and suspected aortoenteric fistula. The patient underwent an emergency surgery, confirming the suspected diagnosis. The surgery performed was the traditionally recommended extraanatomical bypass with aortic ligation and repair of the intestinal defect. We describe the clinical condition and provide an up-to-date overview of diagnosis and treatment by reviewing the literature. We believe the therapeutic decision should be personalized by assessing the anatomy of the aneurysm, the patient’s clinical status, the degree of local contamination, and the surgeon’s experience with each of the techniques.
Aortoenteric fistula (AEF)ecommunication between the aorta and the intestinal lumeneis a very rare and extremely serious condition, which requires emergency treatment in most cases. Despite the different surgical treatment strategies, it still has a very high morbidity and mortality. The AEF may be primary, especially associated with aortic aneurysms, or secondary, more frequent and associated with previous aortic surgery using prosthetic graft. Clinical manifestation is usually gastrointestinal bleeding, sometimes associated with signs of sepsis. Classical treatment with conventional surgery is still in effect, and the aortic ligature and extraanatomical bypass is the most recommended option, although this presents the disadvantage of a high morbidity and mortality associated with it.1e5
Servicio de Angiologı´a y Cirugı´a Vascular, Hospital Universitario Dr. Peset, Valencia, Espa~ na. Correspondence to: Beatriz Genoves-Gasco, MD, Servicio de Angiologı´a y Cirugı´a Vascular, Hospital Universitario Dr. Peset. Avda. Gaspar Aguilar, 90. 46017 Valencia, Espa~ na; E-mail: [email protected] Ann Vasc Surg 2012; 26: 730.e1e730.e5 DOI: 10.1016/j.avsg.2011.11.030 Ó Annals of Vascular Surgery Inc. Published online: April 13, 2012
Conversely, endovascular treatment is a promising option, especially in emergency situations and in patients at high surgical risk owing to its minimally invasive nature.6 It is also especially attractive in case of active or recent hemorrhage and hemodynamic instability.7 Excellent results have been reported in the short-term in many cases,8e10 although in the long-term, the problem of prosthetic graft infection does not seem to be resolved.6,7 In addition, it is not possible to perform endovascular treatment in all patients owing to the anatomical requirements needed for a correct fixation and proper sealing of the endograft.
CLINICAL CASE A 65-year-old man with a personal history of dyslipidemia, chronic obstructive pulmonary disease, and inguinal herniorrhaphy arrived at the emergency department in an ambulance after an episode of abdominal pain and massive rectal bleeding with loss of consciousness. During his stay in the emergency department, he presented with sustained hypotension and an episode of hematemesis. Physical examination revealed a pulsatile abdominal mass, objectifying the existence of an abdominal 730.e1
730.e2 Case reports
aortic aneurysm (AAA) on abdominal ultrasonography. Computed tomography (CT) with intravenous contrast showed a fusiform pararenal AAA with a maximum diameter of 92 mm, which included the left renal artery and extended up to the iliac bifurcation. We also observed a duodenal loop in close contact with its anterior wall, with possible solution of continuity (Fig. 1). With the suspected diagnosis of AEF, the patient underwent an emergency open surgery. We performed an initial axillobifemoral bypass from the right axillary artery using a prosthetic expanded polytetrafluorethylene graft. Subsequently, we accessed the retroperitoneum by means of median laparotomy. First, we proceeded to dissect the aortic neck and both primitive iliac arteries. During the process of detachment of the duodenal loop from the anterior wall of the aneurysm, we discovered the AEF in the third duodenal portion (Fig. 2). We sutured the duodenal wall aneurysm defect and performed the resection of the aneurysm after proximal aortic clamping between the renal arteries. A proximal infrarenal suture of the aneurysm was then performed (double continuous polypropylene suture) and a portion of omentum was wrapped around the closure site. During surgery, the patient remained hemodynamically stable. After surgery, in the intensive care unit, the patient remained sedated, adapted to mechanical ventilation, and started to present hemodynamic instability, requiring vasoactive drugs. Clinical course from the outset showed a slow healing pattern, with gradual multiorgan dysfunction associated with renal, respiratory, and hemodynamic failure. Owing to the unfavorable clinical course and findings on CT of fluid in the peritoneal cavity, dehiscence of the abdominal suture was suspected, and an exploratory laparotomy was performed at 4 days after the admission. Serous fluid was drained, with no macroscopic appearance of an infection, and verifying that intestinal suture presented a good aspect without dehiscence. Subsequently, during his stay in the intensive care unit, the patient developed episodes of nosocomial infection, with Pseudomonas aeruginosa and Acinetobacter baumannii present in the bronchial secretions. Empirical antibiotic therapy was administered in the beginning, and later the therapy was chosen according to antibiogram. On the 12th day, CT again revealed the existence of abdominal fluid, and hence a percutaneous drainage was positioned. After a period with no significant clinical or hemodynamic alterations, the patient presented with progressive worsening on day 27 after the
Annals of Vascular Surgery
Fig. 1. Computed tomography with intravenous contrast. Duodenal loop adhered to the anterior wall of the abdominal aortic aneurysm (AAA).
Fig. 2. Intraoperative photograph of the AAA and aortoduodenal fistula. (1) Duodenal wall defect. (2) Aortic wall defect.
admission. Nonrecoverable cardiac arrest occurred on the 32nd day in a state of distributive shock.
DISCUSSION Primary AEF is a rare condition typically characterized by the clinical triad of gastrointestinal bleeding, abdominal pain, and pulsatile mass, although the complete triad is only present in a minority of patients.1 In most cases, they are associated with aneurysmatic aortas and present as repeated gastrointestinal bleeding. The time interval between the
Vol. 26, No. 5, July 2012
first bleeding and massive bleeding may range from hours to even months. The incidence reported in the literature is between 0.04% and 0.07%.11,12 In the case of primary AEF, there is a communication between the gastrointestinal tract and the aorta, without a history of surgery (we otherwise define this as secondary). Approximately 80% of cases are caused by an AAA, and atherosclerosis is the most common etiology. Other causes of AEF are mycotic or traumatic aneurysms, radiation, neoplasias, peptic ulcers, inflammatory processes such as appendicitis or diverticulitis, cystic medial necrosis, or ingestion of foreign bodies. Microorganisms more commonly responsible for mycotic aneurysms are Salmonella or Klebsiella, although Mycobacterium tuberculosis, Treponema pallidum, fungi, Staphylococcus, and Streptococcus, among others, have also been found.13,14 Although primary AEF can affect any part of the gastrointestinal tract, the duodenum is involved in 82% of cases, especially the third and fourth portion.15 Mortality rates are extremely high, close to 100% in untreated cases or those with active bleeding, and 30% to 40% in cases treated by surgery.11,13,16 Diagnosis is especially difficult because it is a very rare entity; classical clinical symptoms are present in a minority of patients (11%),17 and diagnostic tests are not definitive in a significant percentage of cases. In cases of gastrointestinal bleeding, the first recommended diagnostic test is endoscopy; however, it should be performed in hemodynamically stable patients.13,18,19 This pathology should be suspected in the presence of active bleeding, an ulcer, an erosion with an adherent clot, or a pulsatile extrinsic mass. Endoscopy is also very useful to exclude other causes of bleeding such as gastroduodenal ulcers or esophageal varices and offers therapeutic possibilities,13 although it is only suggestive of AEF in 25% of patients13,18 and is not available urgently at many hospitals. With arteriography, we only find extravasation of contrast within the gastrointestinal tract in 26% of cases; however, this defines arterial anatomy and is useful for aortic reconstruction planning.2,5 Currently, with the progress of CT in terms of speed, quality of the image, and resolution and owing to its greater availability and it being a less invasive diagnostic test, it has been proposed as the test of choice and is being used initially to confirm the suspicion of this entity.13,14 Gastrointestinal bleeding in the presence of a known AAA suggests an AEF, and an emergency CT with intravenous contrast is indicated,11 the diagnostic success rate of which is 60%. Presence of air in the aorta or
Case reports 730.e3
contrast in gastrointestinal tract is a highly suggestive image. For an early diagnosis, a high rate of suspicion is essential, especially in patients with AAA who present with gastrointestinal hemorrhage. In the absence of knowledge of the cause of massive hemorrhage, it is recommended to perform an emergency exploratory laparotomy.18 Monitoring blood pressure with maintenance of systolic pressure between 60 and 100 mm Hg may reduce the risk of recurrent bleeding during the period before surgical repair.11 The three goals of surgery are to control the hemorrhage, to control the infection, and to keep distal perfusion.12 As surgical treatment, extraanatomic bypass is recommended to avoid contamination of the prosthesis and to repair the intestinal defect.13 The in situ graft using a polytetrafluorethylene or polyester prosthesis is suitable in unstable patients and in cases where the level of contamination is low. Another option is reconstruction with cryopreserved grafts, but it has disadvantages such as the possibility of rupture or aneurysm formation, a higher cost,20,21 and difficult availability, especially in emergency situations. A portion of the omentum should be interposed between the vascular anastomosis and the adjacent bowel.11,13 In contrast, endovascular surgery can enable emergency exclusion in an unstable patient using a minimally invasive approach and, together with antibiotics, may be a solution even during long periods. There are reported cases of AEF successfully treated with endovascular exclusion,22e25 and some authors suggest it could be the treatment of choice in cases without signs of sepsis.6,12,24,25 Even then, it is not still considered a definitive treatment, although it may serve as a bridge to the generally recommended procedure ‘‘perform secondary and deferred open surgery.’’ This is because with endovascular treatment, the risk of fulminant sepsis and recurrent bleeding remains owing to a persistent source of untreated infection.7,11,26e28 Furthermore, this needs more strict anatomic requirements than open surgery, and the endograft is not always available on an urgent basis. In our case, the absence of a proximal neck of the aneurysm, which would enable proper proximal fixation of the endograft and therefore performing correct endovascular exclusion, enabled us to rule out this option from the outset (Fig. 3). The absence of the aneurysm’s proximal neck also significantly hinders with performing proximal aortoprosthetic anastomosis and encouraged not performing in situ reconstruction, which is better tolerated hemodynamically and recommended by some authors in
730.e4 Case reports
Annals of Vascular Surgery
diagnosis of AEF should be ruled out first in patients with significant gastrointestinal bleeding and known aortic aneurysm. The discussion among the different surgical options is still open. The decision to be made between these alternatives, including endovascular treatments, should be personalized by assessing a series of variables: the advantages and disadvantages of each one of the techniques, the anatomy of the aneurysm, the patient’s clinical status, the degree of infection or local contamination, and the surgeon’s personal experience. REFERENCES
Fig. 3. Computed tomography with intravenous contrast. Absence of the infrarenal neck of the AAA.
cases of not very significant contamination. Therefore, given the anatomical characteristics, we opted for the classical surgical treatment, that is, aortic ligature and extra-anatomical bypass. Although this is the most recommended treatment and the patient was kept stable at all times during surgery, he began to become hemodynamically unstable during the immediate postoperative period. It is precisely this problemdthe hemodynamic repercussiondthat is the main disadvantage of this surgical option. With regard to the intestinal repair, we opted for primary closure given the limited defect, although local resection may be required in the cases of major defects. The result depends on the delay in diagnosis, the patient’s general condition, the degree of contamination, and the location of the complication in the aorta.17 Furthermore, antibiotic therapy should be initiated since the suspicion of AEF, first empirical against the microorganisms described earlier in the text and subsequently specific according to the result of the culture tests. This has to be maintained for 7 to 10 days if culture results are negative and 4 to 6 weeks if they are positive.13,14 A longer duration of the therapy or even antibiotic treatment for a lifetime is recommended in cases of endovascular exclusion.26 In summary, early diagnosis and surgery are vitally important for the patient’s survival. The
1. Saers SJ, Scheltinga MR. Primary aortoenteric fistula. Br J Surg 2005;92:143e52. 2. Bergqvist D. Arterioenteric fistula. Review of a vascular emergency. Acta Chir Scand 1987;153:81e6. 3. Montgomery RS, Wilson SE. The surgical management of aortoenteric fistulas. Surg Clin North Am 1996;76:1147e57. 4. Busuttil SJ, Goldstone J. Diagnosis and management of aortoenteric fistulas. Semin Vasc Surg 2001;14:302e11. 5. Goshtasby P, Henriksen D, Lynd C, et al. Recurrent aortoenteric fistula: case report and review. Curr Surg 2005;62:638e43. 6. Burks JA Jr, Faries PL, Gravereaux EC, et al. Endovascular repair of bleeding aortoenteric fistulas: a 5-year experience. J Vasc Surg 2001;34:1055e9. 7. Chuter TA, Lukaszewicz GC, Reilly LM, et al. Endovascular repair of a presumed aortoenteric fistula: late failure due to recurrent infection. J Endovasc Ther 2000;7:240e4. 8. Allen RC, Sebastian MG. The role of endovascular techniques in aortoesophageal fistula repair. J Endovasc Ther 2001;8:602e3. 9. Semba CP, Sakai T, Slonim SM, et al. Mycotic aneurysms of the thoracic aorta: repair with use of endovascular stentgrafts. J Vasc Interv Radiol 1998;9:33e40. 10. Leobon B, Roux D, Mugniot A, et al. Endovascular treatment of thoracic aortic fistulas. Ann Thorac Surg 2002;74: 247e9. 11. Chaikof EL, Brewster DC, Dalman RL, et al. The care of patients with an abdominal aortic aneurysm: The Society for Vascular Surgery practice guidelines. J Vasc Surg 2009;50:S2e49. 12. Baril DT, Carroccio A, Ellozy SH, et al. Evolving strategies for the treatment of aortoenteric fistulas. J Vasc Surg 2006;44: 250e7. 13. Lemos DW, Raffetto JD, Moore TC, Menzoian JO. Primary aortoduodenal fistula: a case report and review of the literature. J Vasc Surg 2003;37:686e9. 14. San Norberto-Garcı´a EM, Carrera-Dı´az S, Cenizo-Revuelta N, et al. Fı´stula aortodueodenal primaria: descripci on de un caso y revisi on de la bibliografı´a. Angiologı´a 2007;59:445e9. 15. Farber A, Grigoryants V, Palac DM, et al. Primary aortoduodenal fistula in a patient with a history of intravesical therapy for bladder cancer whith bacillus Calmette-Guerin: review of primary aortoduodenal fistula without abdominal aortic aneurysm. J Vasc Surg 2001;33:868e73. 16. Sweeney MS, Gadacz TR. Primary aortoduodenal fistula: manifestations, diagnosis and treatment. Surgery 1984;96: 492e7. 17. Roche-Nagle G, O’Donnell DH, Brophy DP, Barry MC. Primary aortoenteric fistula. Am J Surg 2008;195:506e7.
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