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Ann Thorac Surg 2002;74:590-591
© 2002 The Society of Thoracic Surgeons

Case report

Aortopulmonary fistula after aortic root replacement

Masahiro Ueno, MD*a, Tatsuya Imada, MDa, Kazuki Nonaka, MDa, Takeshi Oda, MDa

a Department of Cardiovascular Surgery, Omura Municipal Hospital, Cardiovascular Center, Omura, Japan

Accepted for publication April 1, 2002.

* Address reprint requests to Dr Ueno, Department of Cardiovascular Surgery, Omura Municipal Hospital, Cardiovascular Center, 133-22 Kogashima-chou, Omura, Nagasaki 856-8561, Japan
e-mail: masaocvc{at}fsinet.or.jp


   Abstract
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 Abstract
Introduction
 Comment
 References
 
This report describes a rare case of aortopulmonary fistula in pseudoaneurysm of the left coronary ostial button. A 66-year-old woman suddenly developed congestive heart failure 3 years after aortic root replacement for acute type A aortic dissection. The diagnosis of aortopulmonary fistula was confirmed preoperatively by aortography, heart catheterization, and spiral computed tomography. She was discharged in good condition after surgical treatment. This serious complication should be considered in patients who have severe congestive heart failure after aortic root replacement.


   Introduction
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 Abstract
 Introduction
Comment
 References
 
Pseudoaneurysm is a rare and potentially fatal complication of aortic root replacement using composite graft. Treatment may require surgery for anastomotic dehiscence or another aortic root replacement [1, 2]. We present an exceptional case of pseudoaneurysm of the left coronary button complicated by fistulization into the right pulmonary artery following aortic root replacement for acute type A aortic dissection.

A 66-year-old woman was admitted to a local hospital following the sudden onset of dyspnea. Three years before, in our institute, she had aortic root replacement for acute type A aortic dissection. She had systemic edema and a rough systolic murmur maximal at the second left intercostal space. Her blood pressure was 90/60 mmHg and she had sinus tachycardia at 110 beats/min based on an electrocardiogram. At the first operation, both coronary ostia were dissected and reattached to a composite graft using a button technique reinforced with gelatine-resorcine-formol (GRF) glue. The patient demonstrated no clinical stigmata of Marfan’s syndrome, although an aortic wall specimen revealed cystic medial necrosis.

Chest radiogram on the current admission revealed cardiomegaly and pulmonary edema. Transthoracic echocardiography demonstrated good left ventricular function and mild mitral and tricuspid regurgitation. Increased right-sided pressure was present based on a Swan-Ganz catheterization (Baxter Healthcare Corp, Irvine, CA). Specific values were: right atrium, 15 mmHg; right ventricle, 64/15 mmHg; pulmonary artery, 55/23 mmHg; and pulmonary capillary wedge pressure, 19 mmHg. She had good cardiac index of 3.0 L/min/m2. Pulmonary thromboemboli were not observed on contrast-enhanced chest computed tomography. The cause of her severe congestive heart failure could not be identified.

After 1 month of intensive medical therapy, aortography showed a pseudoaneurysm surrounding the left coronary ostia, and contrast medium flowed directly into the right pulmonary artery (Fig 1). An oxygen saturation step-up from the right ventricle to the right pulmonary artery was recorded by cardiac catheterization. A left to right shunt ratio of 50% was calculated. Shunt flow was obtained by spiral computed tomography at a scan delay of 12 seconds after a 100-mL bolus of contrast medium (Fig 2). The patient was then transferred to our institute without further complications.



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  		Fig 1. Aortography revealed pseudoaneurysm surrounding the left coronary ostia (arrow) and contrast medium flowing directly into the right pulmonary artery (r-PA). (LCA = left coronary artery).

 


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  		Fig 2. Chest spiral computed tomography revealed shunt flow that made the defect of contrast medium on the right pulmonary artery (arrow).

 
We operated on her 1 week after admission. Intraoperative transesophageal echocardiography revealed an unidentified space with blood flow around the left coronary ostia. During surgery, deep hypothermic circulatory arrest was introduced to facilitate lysis of the mediastinum and avoid unexpected bleeding from the pseudoaneurysm. In addition, continuous retrograde coronary cardioplegia and selective cerebral perfusion were applied for myocardial and cerebral protection. The ascending aortic graft was resected to preserve the right coronary ostia. Dehiscence at the left coronary ostia was observed. An opening of 15 mm in diameter was found on the right pulmonary artery. No thrombi or fibrous ingrowth were present, either above or below the mechanical valve. The pulmonary artery was repaired using a Bard Sauvage filamentous fabric patch (C.R. Bard, Haverhill, MA). The ascending aorta was replaced using a new 22-mm Dacron (C.R. Bard) tube graft. The left coronary artery was reattached to the aortic graft using an 8-mm Dacron graft positioned on the posterior and right side of the aortic graft.

The postoperative course was uneventful. Postoperative aortography revealed no leakage around the left coronary ostia. Histology of the left coronary artery revealed disruption of the medial elastic fibers, thinning of the intima, and hemosiderin deposition on the adventitia. The patient was discharged from the hospital in good condition 28 days after surgery.


   Comment
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 Abstract
 Introduction
 Comment
References
 
This report describes an exceptional case of fistulization between the coronary button and pulmonary artery. There is no doubt that prompt diagnosis and aggressive treatment of this condition would provide for an excellent prognosis. Aortography is commonly used to detect fistula, and cardiac catheterization confirms the diagnosis [3–5]. In addition, transthoracic or transesophageal echocardiography can be successful in detecting shunt flow [4, 5]. In the present case, spiral computed tomography was also utilized to clarify shunt flow.

The formation of a pseudoaneurysm appeared to be related to the fragility of the dissected coronary ostia. While GRF glue was used to reinforce the button, application of a Teflon felt washer and full-thickness suturing might have prevented complications. On the other hand, redissection associated with GRF glue has been implicated [5, 6]. It is speculated that the cause of redissection might be due to a toxic effect of the formalin component. In our case, the left coronary specimen showed no relationship between left coronary ostial dehiscence and GRF glue. However, the difficulty in applying formalin to a narrowly dissected space around the left coronary artery could cause a surplus or inadequate mixing of the glue components. Therefore, we recommended that GRF glue should be applied with extreme caution to prevent potential necrosis due to formalin.

In summary, aortopulmonary fistula should be considered in patients who have a sudden onset of congestive heart failure after aortic root replacement.


   References
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 Abstract
 Introduction
 Comment
 References
 

  1. Svensson L.G., Crawford E.S. Statistical analysis of operative results. In: Svensson L.G., Crawford E.S., eds. Cardiovascular and vascular disease of the aorta. Philadelphia: Saunders, 1997:432-455.
  2. Aoyagi S., Kosuga K., Akashi H., Oryoji A., Oishi K. Aortic root replacement with a composite graft: results of 69 operations in 66 patients. Ann Thorac Surg 1994;58:1469-1475.[Abstract/Free Full Text]
  3. Piciche M., De Paulis R., Chiariello L. A review of aortopulmonary fistulas in aortic dissection. Ann Thorac Surg 1999;68:1833-1836.[Abstract/Free Full Text]
  4. Chevalier P., Moncada E., Kirkorian G., Touboul P. Acquired aortopulmonary fistula in pseudoaneurysm of the aorta six years after a Bentall operation. J Thorac Cardiovasc Surg 1995;110:1143-1144.[Free Full Text]
  5. Bingley J.A., Gardner M.A.H., Stafford E.G., et al. Late complications of tissue glues in aortic surgery. Ann Thorac Surg 2000;69:1764-1768.[Abstract/Free Full Text]
  6. Kazui T., Washiyama N., Bashar A.H.M., et al. Role of biologic glue repair of proximal aortic dissection in the development of early and midterm redissection of the aortic root. Ann Thorac Surg 2001;72:509-514.[Abstract/Free Full Text]



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