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J Thorac Cardiovasc Surg 2003;126:598-600
© 2003 The American Association for Thoracic Surgery
Brief communication |
a Department of Cardiovascular Surgery, Saitama Medical School, Saitama, Japan
b Department of Cardiology, Saitama Medical School, Saitama, Japan
c Department of Cardiothoracic Surgery, University of Tokyo, Tokyo, Japan
Received for publication November 19, 2002; accepted for publication December 9, 2002.
* Address for reprints: Kazuhito Imanaka, MD, Department of Cardiovascular Surgery, Saitama Medical School, 38 Morohongo, Moroyama-machi, Iruma-gun, Saitama 350-0495, Japan
imanaka{at}saitama-med.ac.jp
A 35-year-old man was referred to our hospital because of persistent fever lasting for 1 month, mild dyspnea, and continuous heart murmur. Oral and intravenous antibiotics had been administered at another hospital. The leukocyte count was 12,000/µL, and the serum C-reactive protein level was 3 mg/dL. The patient had never had severe pain in the chest or in the back, but he had a 1-year history of multiple systemic abscesses that spontaneously developed and healed. Several imaging studies revealed a dissecting aneurysm in the ascending aorta that compressed the pulmonary artery (PA), causing severe pulmonary stenosis (pressure gradient, 75 mm Hg on echocardiography), and that was accompanied by a fistula into the PA (Figure 1). Proximally, the aneurysm reached the level of the aortic valve annulus. A moderate amount of right pleural effusion and marked liver congestion were also noted. On that day, he was admitted to our hospital. However, severe PA stenosis precluded successful right-heart catheterization. Antibiotic therapy was discontinued, but blood culture failed to disclose causative organisms. The clinical condition of the patient remained almost stable for 5 days, but then he suddenly fell into a state of cardiogenic shock and multiple organ failure on the sixth day and underwent emergency surgical intervention. The right ventricle was severely distended and had poor contractility. Intraoperative blood sampling revealed that the Qp/Qs ratio was 4.0. The aneurysm in front of the PA was 5 cm in diameter, and hematoma was absent. The almost round intimal tear in the ascending aorta, 8 mm in diameter, appeared to be a chronic change. The PA wall was gray and very floppy, and there was an irregularly shaped orifice of the fistula, 4 mm in size, just above the sinus of the pulmonary valve (Figure 2). The aortic root and the ascending aorta were replaced with a cryopreserved aortic allograft. The PA wall around the orifice of the fistula was resected, and the PA was reconstructed with the remnant of the aortic allograft. However, in the proximal suture line in the sinus of the pulmonary valve, the fragile PA wall was lacerated, and the resuturing had to incorporate the pulmonary valve and its annulus. The cardiopulmonary bypass was easily discontinued with a moderate amount of inotropic support. Histologically, the resected PA wall was necrotic and had completely lost its normal structure. After the operation, residual pulmonary stenosis (pressure gradient, 40 mm Hg) with mild regurgitation was present and caused persistent right-heart failure. Despite good left ventricular function, central venous pressure remained greater than 15 mm Hg, and multiple organ failure did not resolve. After a nearly 4-week stay in the intensive care unit, the patient underwent PA root replacement with annulus enlargement (from 18 mm to 24 mm in diameter) using a cryopreserved pulmonary allograft. Again, he was easily separated from the cardiopulmonary bypass, but fulminant Candida species–induced mediastinitis developed, and the patient died 3 weeks later. Autopsy revealed no unacceptable structural condition in the heart.
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Aortopulmonary fistula caused by aortic dissection is a rare occurrence. Its cause has not been well documented and tends to differ from case to case.1-3 It can occur either in the acute or chronic phase of aortic dissection, either with or without previous cardiovascular surgery.
In the present case, several difficult problems were present. One was the possible close relationship between the fistula and infection. Judging from the clinical course and the laboratory data, a predisposing infection was strongly suggested. The use of prostheses should be avoided because recurrent infection is fatal. The possibly infectious aneurysm, which had reached the level of the aortic valve annulus, required extensive surgical intervention with an allograft.
Another problem was the severely damaged PA wall. Some surgeons have reported successful closure of a fistula with a pledget-supported suture. We did not use this method because of suspected infection and the severely damaged PA wall. The histologic findings also support our decision. However, the PA wall was extensively abnormal, and the suture line was disrupted. Resuturing incorporating the pulmonary valve annulus caused mild residual stenosis that could not be tolerated in the presence of severe right ventricular dysfunction. Pulmonary root replacement can be a method of choice in such patients. Therefore, we believe that a pulmonary allograft should also be made available whenever possible and should be implanted during the initial operation, if necessary.
The last problem was the timing of the operation. Emergency intervention was required in most reported patients with aortopulmonary fistulae and in this patient also, although the history of the present illness was rather long, and his general condition was fair at first. His severely impaired general condition 6 days later inevitably hampered postoperative recovery. Clearly, the timing of the operation should be decided on a case-by-case basis, but we recommend performing the operation as soon as possible once the diagnosis of aortopulmonary fistula is established.
References
This article has been cited by other articles:
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  M. Mukadam, J. Barraclough, P. Riley, and R. Bonser Acquired aorto-pulmonary artery fistula following proximal aortic surgery Interactive CardioVascular and Thoracic Surgery, October 1, 2005; 4(5): 388 - 390. [Abstract] [Full Text] [PDF]
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