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Asian Cardiovasc Thorac Ann 2003;11:272-274
© 2003 Asia Publishing EXchange Ltd


CASE STUDY

Posttraumatic Intrapancreatic Aortosplenic Fistula Causing Portal Hypertension

Òscar Estrada, MD, Carlos-A Mestres, FETCS1, Miguel-A López-Boado, MD, Juan A Salmerón, MD2, José Visa, PhD, Juan-Carlos García-Valdecasas, PhD

Department of General Surgery
1 Department of Cardiovascular Surgery
2 Department of Hepatology, Hospital Clinic, University of Barcelona, Barcelona, Spain

For reprint information contact: Carlos-A Mestres, FETCS Tel: 34 93 227 5515 Fax: 34 93 451 4898 email: cmestres{at}clinic.ub.es Department of Cardiovascular Surgery, Hospital Clinic, University of Barcelona, Villarroel 170, 08036 Barcelona, Spain.


    ABSTRACT
 TOP
 ABSTRACT
 Introduction
 Case Report
 Discussion
 References
 
Arterioportal fistulas are uncommon. The case of a patient with massive uncontrollable esophageal variceal bleeding is presented. Reversible portal hypertension was caused by a posttraumatic giant intrapancreatic aortosplenic fistula. Percutaneous closure was unsuccessful, and pancreatectomy was performed to control the bleeding. The case is discussed and the literature on this exceptional cause of portal hypertension is reviewed.


    INTRODUCTION
 TOP
 ABSTRACT
 Introduction
 Case Report
 Discussion
 References
 
Arterioportal fistulas are uncommon. They usually develop after liver trauma or gastrohepatic surgery. They can lead to severe upper gastrointestinal bleeding or congestive heart failure. The literature shows only scanty reports on the clinical picture or treatment. Here we describe the case of a patient presenting with intractable upper gastrointestinal bleeding secondary to portal hypertension caused by an intrapancreatic giant aortosplenic fistula 1 year after repair of an abdominal stab wound.


    CASE REPORT
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 ABSTRACT
 Introduction
 Case Report
 Discussion
 References
 
A 33-year-old male was transferred from another center because of uncontrollable esophageal bleeding. His past medical history included laparotomy for an abdominal stab wound 1 year earlier. The surgical report referred only to a small bowel lesion and pancreatic exploration that produced no abnormal findings. The patient was an active smoker and intravenous heroin addict with positive serology for the hepatitis C virus (HCV). No liver biopsy was previously performed, although biochemical analysis suggested liver dysfunction due to the HCV infection. Two months before admission, the patient had an episode of gastrointestinal bleeding with melena and required packed red cell transfusion. No esophageal varices were found on gastroscopy then.

Upon this admission, the patient was found to be in shock because of uncontrollable esophageal hemorrhage. Gastroscopy showed 3 large esophageal varices. Somatostatin was started. Seven hours later, recurrent severe bleeding required Sengstaken-Blakemore balloon tamponade. Inadequate control of bleeding prompted an emergency exploratory laparotomy to insert a portosystemic shunt. Through a bilateral subcostal approach, the liver surprisingly was found to be of normal gross appearance. The decision was made to close the wound and proceed with angiographic study.

Angiography showed a large fistula between the abdominal aorta and the splenic vein (Figure 1Go). The origin of the fistula was located between the celiac axis and the superior mesenteric artery. Every attempt at closure with a coil failed because the large flow of the fistula caused the coil to migrate, and only temporary control was achieved by inserting an angioplasty balloon. With the bleeding under control, the condition of the patient was stabilized. The following day, a second laparotomy was performed. Severe inflammatory reaction was found, making organ and vascular dissection difficult. The aorta was approached superiorly from the celiac axis and distally from the renal arteries, and the pancreas was dissected. The left renal vein was ligated to expose the pancreatic tail and the suprarenal aorta. A large fistula (2 cm) was found to arise from the anterior aspect of the aorta above the superior mesenteric artery and entering the body of the pancreas, where a heavy mass was palpated. A two-third pancreatic resection including the tail and part of the body was finally performed under stapling. Upon resection of the fistula, the balloon catheter was seen coming out of the aorta (Figure 2Go). The origin of the fistula was resected and closed with a double 3/0 polypropylene suture. The abdomen was closed in the routine fashion, leaving 2 large-bore drains in place.



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Figure 1. Arteriography showing the communication between the aorta and the portal axis.

 



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Figure 2. (A) Operative view of the fistula under clamping. (B) Pathological specimen.

 
The patient had an uneventful postoperative course with cessation of bleeding. Serology confirmed HCV infection with a negative result for the human immunodeficiency virus. Liver biopsy showed a normal liver with minimal inflammation and foci of lipoid changes. He was transferred to the referring center.

Five months after the operation, he was doing well and leading an active life, although there were doubts about the control of his addiction.


    DISCUSSION
 TOP
 ABSTRACT
 Introduction
 Case Report
 Discussion
 References
 
Arterioportal fistulas are an exceptional cause of portal hypertension. More than 90% of the cases of gastroesophageal variceal bleeding are due to cirrhosis. The remaining 10% are due to an increase in the resistance of the portal circulation, venoclusive disease, or even constrictive pericarditis.1 In only a handful of patients is an increase in portal flow considered as the cause. Arteriovenous fistulas are usually congenital or acquired before abdominal surgery. Often they are associated with hepatocarcinoma, abdominal trauma, or hepatic artery aneurysm.2 Most of them are intrahepatic and very few are extrahepatic.3–5 In these patients, hyperflow and secondary liver fibrosis can induce portal hypertension. Liver biopsy is considered the most common origin of iatrogenic arterioportal fistula;6 however, most of the cases are asymptomatic and the fistula tends to close spontaneously. The time between shunt formation and onset of symptoms may vary between a few days and up to 48 hours, according to literature reports.7 The common clinical picture is portal hypertension with abdominal pain and severe bleeding. A triad has been proposed to depict symptomatic arterioportal fistulas: upper abdominal thrill, portal hypertension, and borderline liver enzyme elevation.8

In the case described here, there was a traumatic extrahepatic fistula. This patient presented with massive upper gastrointestinal bleeding. No abdominal thrill was found on palpation. Ascitis, esophageal varices, and splenomegaly were also present. However, an initial exploratory laparotomy and liver biopsy failed to confirm a cirrhotic liver. In such cases, angiography is the main diagnostic tool. It is most helpful at the time of embolization, especially of intrahepatic shunts with peripheral flow due to a lower rate of spontaneous occlusion. There is no consensus as regards the effectiveness of transcutaneous intrahepatic portosystemic shunt. Although there are a few successful reported cases, some major complications have occurred, such as right heart failure because of hyperflow. We found no cases of spontaneous closure of extrahepatic shunts in the English literature, and surgery is the treatment of choice if complete occlusion cannot be achieved with angiographic embolization.

In our case, the initial diagnosis of noncirrhotic portal hypertension was followed with temporary balloon closure of the fistula. That allowed stabilization of the patient before surgical treatment. This case confirms the importance of arterioportal fistula as part of the differential diagnosis of patients with upper gastroesophageal variceal bleeding without a history of liver disease. A multipronged approach in patients with massive esophageal bleeding of unclear etiology is mandatory, even in exceptional cases as the one presented here.


    REFERENCES
 TOP
 ABSTRACT
 Introduction
 Case Report
 Discussion
 References
 

  1. MacMathuna P, Westaby D. Portal hypertension: pathophysiology, diagnosis and treatment. In: Millward-Sadler GH, Wright R, Arthur MJP, editors. Wright’s liver and biliary disease. London: Saunders, 1992:1296–334.

  2. Aithal GP, Alabdi BJ, Rose JD, James OF, Hudson M. Portal hypertension secondary to arterio-portal fistulae: two unusual cases. Liver 1999;19:343–7.[Medline]

  3. Salvat F, Burelle H, Bohe D, Boyer F. Post-traumatic extrahepatic arterioportal fistula. A case with a spontaneously favourable course [French]. J Radiol 1980;61:185–7.[Medline]

  4. Hung CF, Tseng JH, Lui KW, Wan YL, Tsai CC, Shem CH, et al. Intractable oesophageal variceal bleeding caused by splenic arteriovenous fistula: treatment by transcatheter arterial embolization. Postgrad Med J 1999;75:355–7.[Abstract/Free Full Text]

  5. Regent D, Hodez C, Bigard MA, Regent MC, Gaucher P, Roussel J. Splenic arterial aneurysm rupturing into the splenic vein. A rare cause of acute portal hypertension in the post-partum period [French]. J Radiol Electrol Med Nucl 1977;58:151–4.[Medline]

  6. Okuda K, Musha H, Nakajima Y, Takayasu K, Suzuki Y, Morita M, et al. Frequency of intrahepatic arteriovenous fistula as a sequela to percutaneous needle puncture of the liver. Gastroenterology 1978;74:1204–7.[Medline]

  7. Redmond PL, Kumpe DA. Embolization of an intrahepatic arterioportal fistula: case report and review of the literature. Cardiovasc Intervent Radiol 1988;11:274–7.[Medline]

  8. Hirner A, Haring R, Bost H, Sorensen R. Hyperkinetic portal hypertension. Arterioportal fistula: problems–case reports–review of the literature [German]. Chirurg 1978;49:303–10.[Medline]





This Article
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Right arrow Articles by Estrada, O.
Right arrow Articles by García-Valdecasas, J.-C.


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