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Ann Thorac Surg 2002;73:1634-1636
© 2002 The Society of Thoracic Surgeons


Case report

Aortopulmonary fistula after coarctation repair with dacron patch aortoplasty

Leo O. Heikkinen, MD, PhD*a, Antero A. Järvinen, MD, PhDa

a Department of Cardiothoracic Surgery, Helsinki University Hospital, Helsinki, Finland

Accepted for publication August 24, 2001.

* Address reprint requests to Dr. Heikkinen, Department of Cardiothoracic Surgery, Helsinki University Hospital, Haartmaninkatu 4, PO Box 340, FIN-00029, Helsinki, Finland
e-mail: leo.heikkinen{at}hus.fi


    Abstract
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 Abstract
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 Comment
 References
 
Fistulous communication between the aorta and the pulmonary parenchyma developed in a 38-year-old woman 19 years after repair of a congenital aortic coarctation with Dacron patch aortoplasty. The fistula, inducing intermittent hemoptysis, arose from the suture line between the prosthetic fabric and the aorta. There was no infectious background or aneurysm at the primary repair site. The aortic segment including the prosthetic patch was resected and replaced with a Dacron tubular vascular prosthesis.


    Introduction
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A Dacron patch aortoplasty technique was earlier widely accepted as a method of aortic coarctation repair. Widening of the aortic isthmus with a rigid, diamond-shaped prosthetic graft has led to many unexpected problems, the great majority related to aneurysm or false aneurysm formation at the repair site even years after primary repair [13].

Development of a fistulous communication between the aorta and the tracheobronchial tree has been a rare occurrence in this connection, with only a few comparable complications documented [48]. Recently Milano and coworkers [6] reported such a fistula developing after patch aortoplasty after chest trauma. All fistulas described have occurred with other coexistent disease, eg, infection, aneurysm, or pseudoaneurysm, at the repair site. However, in our earlier reports no patient simultaneously had aortobronchial or aortopulmonary fistula related to an aneurysm after Dacron patch aortoplasty [1]. Herein we describe a female patient with an aortopulmonary fistula discovered at the aortoplasty site, which developed without a coexistent aneurysm 19 years after Dacron patch isthmusplasty.

A 38-year-old woman was admitted on an emergency basis because of undefined symptoms of hemoptysis or hematemesis. At age 19 she had undergone aortic coarctation repair with the technique of Dacron onlay patch aortoplasty. Owing to recent excessive consumption of alcohol, her gastrointestinal tract was considered as the primary origin of bleeding. Her hemoglobin level was 9.8 g/dL, she had no fever, and her C-reactive protein level was less than 5 mg/L. Examination of the upper gastrointestinal tract proved negative. At bronchoscopy blood was evident at the level of the left main bronchus.

A routine chest roentgenograph showed no abnormality, no aneurysm, and no infiltration or consolidation of the lung. An aortogram demonstrated only slight irregularity in the wall of the descending thoracic aorta distal to the left subclavian artery. No aneurysm, true or false, or any fistulous communication was observed. Computed tomographic scanning revealed a mass of soft tissue density (2 cm x 4 cm), suggesting a hematoma adjacent to the Dacron patch at the level of the left lower lobe bronchus. Past history, findings in computed tomography, and clinical status together raised suspicion of the existence of a fistulous communication between the aorta and left lung at the level of the prosthetic patch.

Prophylactic antimicrobic therapy was given with vancomycin 500 mg and imipenem 1 g four times daily for 1 week to prevent infection of the prosthetic material for further aortic reconstruction. The left thoracic cavity was entered through the previous thoracotomy route. The left lung was strongly adhered to the thoracic wall and firmly fixed on the descending thoracic aorta against the prosthetic fabric.

Under femorofemoral bypass, the aortic arch was clamped between the left carotid and subclavian arteries. The descending thoracic aorta was closed distal to the former aortoplasty. Longitudinal aortotomy revealed a round hole 5 mm in diameter located at the distal end of the anastomosis between the Dacron patch and the aorta (Fig 1). This hole communicated directly with the peripheral pulmonary parenchyma. No traces of macroscopic infection were visible. Specimens for bacterial and fungal cultures were taken from around the hole.



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Fig 1. The removed Dacron patch, with a round hole 5 mm in diameter (arrow) visible in the anastomotic suture line between prosthetic graft and aortic wall.

 
The whole aortic segment with the prosthetic patch was removed, and the continuity of the aorta was restored with a gelatin-coated tubular Dacron vascular prosthesis 20 mm in diameter. The new prosthesis was carefully covered with a parietal pleural flap. No special procedure was needed for the pulmonary side of the fistula.

Fibrosis was evident histologically. A neointimal layer corresponding to the lumen covered the Dacron graft. The other side was covered with connective tissue and partly with columnar epithelium originating from pulmonary tissue. Small amounts of inflammatory cells were detected, but otherwise the microscopic examination revealed no signs of infection. Bacterial and fungal cultures were negative.

Antibiotics were continued with ofloxacin 200 mg twice daily for 4 weeks. Recovery was uneventful, and the patient was discharged 12 days postoperatively. At follow-up 54 months later she had had no recurrent episodes of hemoptysis and was doing well overall.


    Comment
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 Comment
 References
 
Intermittent hemoptysis is a pathognomonic feature of an aortobronchopulmonary fistula. Any history of thoracic aortic operation in a patient coughing up blood should promptly direct the diagnostic methods to the possibility of such a fistula.

A standard chest roentgenogram may detect an aneurysm or an unspecific pulmonary density or may reveal no abnormalities [48]. Direct visualization of the fistulous communication has rarely been possible, although Caes and associates [4] succeeded in demonstrating a fistulous tract with magnetic resonance imaging technique. Computed tomography has provided additional information in differentiating the aneurysm from soft tissue density, infiltrate, or hematoma as signs of bleeding [47]. Conventional aortography can identify any aneurysm or a false aneurysm, but is also of great importance in evaluating the postoperative results, eg, residual stenosis after coarctation repair [5, 7, 8].

Bronchoscopy and preoperative respiratory function studies should be considered carefully if an aortobronchopulmonary communication is suspected, even more so if the quantity of expectorated blood is abundant. Bronchoscopic examination can be both hazardous and dangerous, and interpretation may be difficult if hemoptysis is massive. Increase in pressure may open a temporarily closed connection between aorta and tracheobronchial tree, provoking a dramatic change for the worse. However, a direct bronchoscope view can reveal the origin of the blood, as in our case.

Microbial origin has been reported to cause this process [5, 8]; we considered an infectious cause for this fistulous tract formation, but the primary coarctation repair and hospital stay 19 years earlier was uneventful, and on readmission, stains and cultures were negative for microbial origin, and microscopic examination also excluded any infectious cause.

Fistulas between the aorta and airways are rare but not unforeseen complications after coarctation repair by Dacron patch onlay technique. So far, all fistulas described between aorta and left tracheobronchial tree have simultaneously existed with an aneurysm or a false aneurysm at the repair site [48]. Referring to our previous findings, the aneurysms have been true ones [9]. The thin and weakened wall of the aneurysm predisposes to and facilitates the erosion process that may result in fistulous tract formation, a process occurring even more easily if the suture line between the aorta and the prosthesis has dehisced. Although we have found an exceptionally large number of aneurysms after isthmic coarctation repair with the Dacron patch onlay technique, we have seen no other simultaneous aortopulmonary communication. In this case, the fistula formed a round hole in the anastomotic suture line.

Undiagnosed aortopulmonary communication is lethal. Endovascular repair can be considered, provided that infection is excluded—otherwise the treatment is surgical. Ultimately, treatment must be started on the basis of clinical symptoms and status if diagnostic imaging methods fail to confirm directly or indirectly fistulous communication.


    References
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 Abstract
 Introduction
 Comment
 References
 

  1. Ala-Kulju K., Heikkinen L. Aneurysms after patch graft aortoplasty for coarctation of the aorta: long-term results of surgical management. Ann Thorac Surg 1989;47:853-856.[Abstract/Free Full Text]
  2. Hehrlein F.W., Mulch J., Rautenburg H.W., et al. Incidence and pathogenesis of late aneurysms after patch graft aortoplasty for coarctation. J Thorac Cardiovasc Surg 1986;92:226-230.[Abstract]
  3. McGoldrick J.P., Brown I.W., Ross D.N. Coarctation of aorta: late aneurysm formation with Dacron onlay patch grafting. Ann Thorac Surg 1988;45:89-90.[Abstract/Free Full Text]
  4. Caes F., Taeymans Y., Van Nooten G. Aortobronchial fistula: a late complication of coarctation repair by patch aortoplasty. Thorac Cardiovasc Surgeon 1993;41:80-82.[Medline]
  5. Favre J.P., Gournier J.P., Adham M., Rosset E., Barral X. Aortobronchial fistula: report of three cases and review of the literature. Surgery 1994;115:264-270.[Medline]
  6. Milano A., De Carlo M., Mussi A., Falaschi F., Bortolotti U. Aortobronchial fistula after coarctation repair and blunt chest trauma. Ann Thorac Surg 1999;67:539-541.[Abstract/Free Full Text]
  7. Miller J.P., Cammarata S.K. Massive hemoptysis 17 years after repair of aortic coarctation. Chest 1994;105:1249-1250.[Medline]
  8. Singh A.K., Reimer R. Subclavian flap repair after mycotic aneurysm of patch repair of coarctation. J Thorac Cardiovasc Surg 1982;84:911-916.[Medline]
  9. Heikkinen L., Sariola H., Salo J.A., Ala-Kulju K. Morphological and histopathological aspects of aneurysms after patch aortoplasty for coarctation. Ann Thorac Surg 1990;50:946-948.[Abstract/Free Full Text]



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This Article
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