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Ann Thorac Surg 2001;71:1363-1365
© 2001 The Society of Thoracic Surgeons

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Case report

Mycotic arch aneurysm and aortoesophageal fistula in a patient with melioidosis

^a Division of Cardiothoracic Surgery, Fletcher Allen Health Care and the University of Vermont, Burlington, Vermont, USA
^b Division of Gastroenterology, Fletcher Allen Health Care and the University of Vermont, Burlington, Vermont, USA
^c Division of Interventional Radiology, Fletcher Allen Health Care and the University of Vermont, Burlington, Vermont, USA

Accepted for publication May 24, 2000.

Address reprint requests to Dr Schmoker, Fletcher 4, 111 Colchester Ave, Burlington, VT 05401
e-mail: joseph.schmoker{at}vtmednet.org

	Abstract

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Aortoesophageal fistula due to an aortic arch aneurysm is a rare entity with an extremely high mortality. There are few reports of successfully managed cases and even fewer of long term survival. We report a case of an aortoesophageal fistula resulting from a mycotic pseudoaneurysm of the distal aortic arch in a patient with melioidosis, its surgical management, and outcome.

	Introduction

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Mycotic aneurysms of the aortic arch are rare, and those resulting in aortoesophageal fistula (AEF) are reportable. We present a case of an AEF from a mycotic aortic arch aneurysm caused by a rare tropical disease.

A 66-year-old Vietnamese male was admitted to our institution with lower chest and epigastric pain, fever, and a single episode of hematemesis. His past medical history was pertinent for splenectomy one month before admission, secondary to multiple splenic abscesses from Pseudomonas pseudomallei. Before his splenectomy, he had an esophagogastroduodenoscopy (EGD) which showed a non-bleeding antral ulcer. This was attributed to use of nonsteroidal anti-inflammatory drugs. The remainder of his past medical history was remarkable for malaria and a positive purified protein derivative.

At initial presentation his blood pressure was 80/50 mm Hg. After successful fluid resuscitation and nasogastric tube lavage, he was admitted to the medical intensive care unit. After several hours, he had another episode of hematemesis and hypotension. EGD revealed a large blood clot in the fundus of the stomach, which could not be completely removed with lavage. There was no ongoing bleeding noted.

Later that evening, the patient suffered another episode of hypotension and hematemesis, leading to crash intubation for airway protection. After gastric lavage, a repeat EGD was performed. Denuded esophageal mucosa was noted in the upper 1/3 of the esophagus, and was presumed secondary to trauma during the difficult airway intubation. A suspected vascular lesion was observed in the greater curve of the stomach and was treated endoscopically. Again, no active bleeding was noted.

At 24 hours after admission the patient suffered an episode of exsanguinating hematemesis. A third EGD revealed a large amount of bright red blood within the esophageal lumen, raising the suspicion of an AEF. To temporize the bleeding by tamponade, an esophageal balloon was placed [Minnesota Four Lumen Tube (MT); Bard, Covington, GA]. The patient was taken emergently to the angiography suite where an aortic arch angiogram revealed an aneurysm (Fig 1). Passage of contrast into the esophageal lumen occurred when the MT balloon was partially deflated, confirming the diagnosis of an AEF. Cardiothoracic surgery was consulted.

View larger version (142K): [in this window] [in a new window]   Fig 1. Aortic arch angiogram showing the distal arch pseudoaneurysm and the esophageal lumen containing contrast. The Minnesota Tube balloon is partially deflated. The arrow points to the aortoesophageal fistula.

The patient was taken emergently to the operating room and prepared for left thoracotomy. Femoral arterial and venous cannulation were immediately performed after full heparinization, cardiopulmonary bypass (CPB) was instituted, and left thoracotomy was undertaken while rapidly cooling the patient. Upon entering the chest, an inflammatory reaction was noted around the aortic arch, which prevented access to the lessor curve. The patient was cooled to 18°C, placed in the Trendelenburg position, and perfusion was stopped. The anterolateral wall of the aortic arch was opened to reveal a 4 cm pseudoaneurysm involving the lesser curve. The aneurysm contained fresh clot, and its posterior wall connected directly to the esophageal lumen. The lesser curve of the aortic arch was debrided back to healthy tissue and reconstructed with a 8 x 5 cm Hemashield patch (homograft was not available). Perfusion was reinstituted with controlled warming. During this time, the esophageal lumen was stapled closed distal to the defect and two 36F. chest tubes were placed. The CPB time was 121 minutes and the circulatory arrest time was 31 minutes. After closure of the thoracotomy incision, the patient was prepared for cervical esophagostomy and open gastrostomy tube placement for completion of esophageal exclusion. The pseudoaneurysm wall, by histologic exam, was found to consist of laminated fibrin and red cell thrombus. Cultures of the aortic wall grew rare Pseudomonas pseudomallei.

Postoperatively the patient was hemodynamically stable. Acute renal failure, which resolved completely after one week, and acute respiratory failure requiring tracheostomy complicated his course. His tracheostomy tube was eventually removed and he was transferred to the rehabilitation unit 43 days after surgery. He subsequently fully recovered and was discharged to await esophageal reconstruction by colonic interposition. He was continued on long term suppressive antibiotics.

	Comment

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Aortoesophageal fistulas are rare, comprising 10% of all aortoenteric fistulas. Most AEF are due to descending thoracic aortic aneurysms and are associated with a very high mortality [1]. The first successful repair was recorded in 1983 [2], but has been followed by few long term surgical successes [3].

To our knowledge, this is the first successfully treated case of AEF complicating a mycotic aortic arch aneurysm. The infectious agent was the bacterium Pseudomonas pseudomallei, found in stagnant tropical water and soil. It causes the rare infectious tropical disease Melioidosis [4], which can remain dormant for years. Our patient was a former rice farmer from Vietnam who recently immigrated to the United States, and probably contracted the bacterium from an open sore or cut. The more localized form of the disease, initially involving the spleen, characterized our patient’s course. There are few reported cases of vascular involvement [5, 6].

Because of the rare occurrence of AEF, the diagnosis is often one of exclusion, and requires a high index of suspicion. Although our patient presented with Chiari’s triad—midthoracic pain, sentinel hemorrhage, and after a symptom free period, final exsanguination [1]—the diagnosis was delayed given his previous history of a gastric ulcer and several inconclusive EGD’s. In retrospect, the diagnosis was initially missed when abnormal upper esophageal mucosa, a consistent finding with AEF, was interpreted as denudation secondary to trauma from attempts at endotracheal intubation. Once suspected, the bleeding was temporized with the use of a MT until the diagnosis was confirmed by angiography.

The operative management of this patient initially consisted of immediate resuscitation on CPB, given that he was almost impossible to ventilate secondary to the pulmonary effects of massive blood transfusions. Profound hypothermic circulatory arrest was necessary given the location of the aneurysm, and because the aortic arch could not be mobilized secondary to the surrounding severe inflammation. Clearly, homograft material would have been most optimal for repair had it been available. The esophageal perforation was managed by exclusion, given the large defect, its location, and the poor overall status of the patient. We could not risk breakdown of a primary repair given the contiguous placement of prosthetic material. A CT scan of the chest performed at the time of discharge corroborated clinical evidence that there was no continuing sepsis associated with the prosthetic material. It will remain to be seen how this patient fares on long term suppressive antibiotics with prosthetic material in place.

	References

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1. Hollander J.E. Aortoesophageal fistula: a comprehensive review of the literature. Am J Med 1991;91:279-287.[Medline]
2. Snyder D.M., Crawford E.S. Successful treatment of primary aorta-esophageal fistula resulting from aneurysm. J Thorac Cardiovasc Surg 1983;85:457-463.[Abstract]
3. Goto H., Utoh J., Hongoh H., et al. Successful treatment of aortoesophageal fistula resulting from aneurysm of the aortic arch. J Cardiovasc Surg 1998;39:425-427.[Medline]
4. Leelarasamee A., Bovornkitti S. Melioidosis: review and update. Rev Infect Dis 1989;11:413-425.[Medline]
5. Torrens J.K., McWhinney P.H.M., Tompkins D.S. A deadly thorn: a case of imported melioidosis. Lancet 1999;353:1016.[Medline]
6. Steinmetz I., Stosiek P., Hergenrother D., Bar W. Melioidosis causing a mycotic aneurysm. Lancet 1996;347:1564-1565.[Medline]

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				J. G. H. Low, A. M. L. Quek, Y. K. Sin, and B. S. P. Ang Mycotic Aneurysm Due to Burkholderia pseudomallei Infection: Case Reports and Literature Review Clinical Infectious Diseases, January 1, 2005; 40(1): 193 - 198. [Abstract] [Full Text] [PDF]

This Article Abstract Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): Joseph D. Schmoker Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Patel, M. A. Articles by D’Agostino, R. Search for Related Content PubMed PubMed Citation Articles by Patel, M. A. Articles by D’Agostino, R. Related Collections Great vessels Esophagus - other