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Ann Thorac Surg 1999;67:539-541
© 1999 The Society of Thoracic Surgeons

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Case Reports

Aortobronchial fistula after coarctation repair and blunt chest trauma

^a Division of Cardiac Surgery, Department of Cardiology, Angiology and Pneumology, University of Pisa Medical School, Pisa, Italy
^b Division of Thoracic Surgery, Department of Surgery, University of Pisa Medical School, Pisa, Italy
^c Department of Radiology, University of Pisa Medical School, Pisa, Italy

Accepted for publication July 16, 1998.

Address reprint requests to Dr Bortolotti, U.O. Cardiochirurgia, Ospedale Cisanello, Via Paradisa 2, 56124 Pisa, Italy
e-mail: u.bortolotti{at}cardchir.med.unipi.it

	Abstract

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A 34-year-old man had development of an aortobronchial fistula 17 years after patch aortoplasty for correction of aortic coarctation and 5 years after blunt chest trauma, an unusual combination of predisposing factors. The clinical presentation, characterized by dysphonia and recurrent hemoptysis, and the surgical findings suggested the posttraumatic origin of the fistula, which was successfully managed by aortic resection and graft interposition under simple aortic cross-clamping, associated with partial pulmonary lobectomy. When hemoptysis occurs in a patient with a history of an aortic thoracic procedure, the presence of an aortobronchial fistula should be suspected. Early diagnosis offers the only possibility of recovery through a lifesaving surgical procedure.

	Introduction

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Aortobronchial fistula (ABF) is a rare condition that is usually fatal if untreated. Chronic thoracic aortic aneurysms have been reported to be the most common cause of ABF [1]. More recently, the occurrence of a fistulous communication between the thoracic aorta and the tracheobronchial tree has been identified as a potential complication of graft replacement of the thoracic aorta [2, 3]. However, ABF complicating correction of aortic coarctation [3–6] or developing from posttraumatic thoracic pseudoaneurysm [7] has seldom been reported. We present the case of a patient who was seen with an ABF late after aortic coarctation repair by patch aortoplasty and after blunt chest trauma with pseudoaneurysm formation, a combination of predisposing factors that we have not seen reported previously.

A 34-year-old man was referred to our hospital in October 1997 because of recurrent episodes of hemoptysis during the 3 months prior to admission. At the age of 17 years, he had undergone repair of aortic coarctation by Gore-Tex patch aortoplasty at our institution. For the next 12 years, the patient remained asymptomatic, although arterial hypertension developed. At the age of 29 years, he sustained blunt trauma to the left hemithorax with multiple rib fractures and pulmonary contusions and facial trauma in a car accident, without apparent sequelae. In April 1997, progressive dysphonia appeared, but a routine chest roentgenogram was not diagnostic. Three months later, he began to experience severe nocturnal hemoptysis with increasing frequency. He was hospitalized repeatedly but no conclusive diagnosis was reached until a pulmonary lesion on the left upper lobe was suggested by the chest roentgenogram. He was transferred to our department in October 1997.

On admission, the patient was in stable condition, with a heart rate of 80 bpm, a respiratory rate of 20/min, and a blood pressure of 120/80 mm Hg. Femoral pulses were evident, and the physical examination was otherwise unremarkable. Routine laboratory findings revealed only moderate anemia (hemoglobin level, 12.0 g/dL). The standard chest roentgenogram showed a small radiopaque area adjacent to the aortic knob, with minimal interstitial infiltration of the left upper lobe (Fig 1). A chest computed tomographic scan demonstrated a pseudoaneurysm at the level of the aortic patch and an area of consolidated parenchyma in the left upper lobe (Fig 2). The sagittal reconstruction of the thoracic computed tomographic scan demonstrated a hematoma in the left upper lobe with obstruction of the segmental bronchus (Fig 3). The presence of an ABF was confirmed by chest magnetic resonance imaging, even though a fistulous connection between the pseudoaneurysm and the tracheobronchial tree could not be visualized.

View larger version (152K): [in this window] [in a new window]   Fig 1. Chest roentgenogram showing a limited radiopaque area around the aortic knob with signs of minimal interstitial infiltration of the left upper lobe.

View larger version (102K): [in this window] [in a new window]   Fig 2. Computed tomographic scan of the chest revealing the presence of a pseudoaneurysm at the level of the patch used for repair of aortic coarctation.

View larger version (138K): [in this window] [in a new window]   Fig 3. Sagittal reconstruction of computed tomographic scan; the left bronchial tree can be recognized (white asterisk). A hematoma is evident in the left upper lobe (black asterisk), with obstruction of a segmental bronchus (white arrowhead).

After administration of heparin sodium, the aortic arch distal to the left carotid artery, the left subclavian artery, and the descending aorta were clamped. The pseudoaneurysm was opened, thus exposing the aortoplasty patch. It was partially detached along its upper circumference from the fibrotic aortic wall, which appeared to be torn, but there was no evidence of suture disruption. Fresh hemorrhagic infiltration of the pulmonary parenchyma near the patch was observed, but the fistulous communication between the thoracic aorta and the tracheobronchial tree was not clearly identified. The pseudoaneurysm was excised, and continuity between the aortic arch and the descending aorta was reconstructed by interposition of an 18-mm tubular Dacron graft. The aortic cross-clamp was released after 22 minutes, and hemostasis was accurately controlled. Subsequently, part of the upper lobe of the left lung, including the Gore-Tex patch and a portion of the aortic wall, was removed with the aid of staples. At the end of the procedure, the new graft was isolated from the lung by covering it with a patch of glutaraldehyde-tanned bovine pericardium.

The patient was extubated 3 hours after operation and discharged to the ward the next day. The postoperative period was complicated by a pulmonary infection caused by Aspergillus fumigatus. The patient was discharged after 3 weeks and at the last follow-up visit, 8 months after reoperation, he had no signs of infection but still complained of moderate dysphonia.

	Comment

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Aortobronchial fistula is a rare condition with a high mortality if untreated [1, 2]. The fistula usually results from erosion of the pulmonary parenchyma or bronchial wall from an adherent aneurysm, which can be of infectious, atherosclerotic, or traumatic origin [1, 3]. Recently, ABF has also been recognized to be a complication of surgical procedures on the thoracic aorta, particularly after graft replacement because of chronic aneurysms [3]. Development of ABF has been observed after repair of aortic coarctation, but to date, only a few such cases have been reported [3–6]. Aortobronchial fistula after patch aortoplasty or graft replacement can be due to false aneurysm formation from a failed anastomotic suture line or from infection of the prosthesis, or it can be the result of erosion of the airways by direct contact between an aortic prosthetic graft and the lung [1, 3].

The initial clinical presentation of ABF is usually characterized by signs of airway compression, such as dyspnea and cough, followed by recurrent episodes of hemoptysis, which can at times be massive and fatal [1, 7]. In our patient, the onset of dysphonia, most likely due to compression or infiltration of the recurrent laryngeal nerve, with subsequent episodes of hemoptysis occurring 17 years after patch repair of aortic coarctation and 5 years after blunt chest trauma indicates that most likely the ABF was related to the unusual combination of such predisposing factors.

Hemoptysis is a nonspecific symptom, but when it occurs in patients with a past history of aortic thoracic procedures, it should raise a high level of suspicion of an ABF [3]. Chest roentgenograms often can be nondiagnostic, as in our patient, because a pseudoaneurysm of the thoracic aorta can be clearly visualized in only 46% of patients [8]. In most instances, correct diagnosis can be achieved only with a computed tomographic scan, which in our patient identified a periaortic hematoma around the aortoplasty patch and a hematoma in the left upper lobe obstructing a segmental bronchus. When the computed tomographic scan reveals both the aortic lesion and secondary changes in the adjacent lung, demonstration of a fistulous communication is not mandatory for a diagnosis of ABF [3].

Once the diagnosis of ABF is confirmed, the only therapeutic option is operation. It is usually carried out on an emergency basis, with an overall early survival rate, in the reported cases, of approximately 75% [3]. Various surgical techniques have been described for the treatment of ABF. The bronchial defect can simply be sutured or managed by a more extensive pulmonary resection [3]. The aortic side of the fistula can be closed by direct suture, with a patch, or by interposition of a prosthetic graft. If the last is indicated, the use of cardiopulmonary bypass or a femoral-atrial shunt should be considered, depending on site of the aneurysm and the condition of the patient [1, 3]. Because the extension of the pseudoaneurysm was limited in our patient, we preferred to correct the lesion by simple aortic cross-clamping. Our patient was treated by standard aneurysmectomy and graft interposition, with excision of all prosthetic material, pseudoaneurysm wall, and infiltrated pulmonary parenchyma. To further avoid the potential risk of secondary infection of the new graft, the latter was isolated from the lung by means of xenograft pericardium.

	References

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1. MacIntosh E.L., Parrott J.C.W., Unruh H.W. Fistulas between the aorta and tracheobronchial tree. Ann Thorac Surg 1991;51:515-519.[Abstract]
2. Ishizaki Y., Tada Y., Takagi A., et al. Aortobronchial fistula after an aortic operation. Ann Thorac Surg 1990;50:975-977.[Abstract]
3. 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]
4. Garniek A., Morag B., Schmahmann S., Rubinstein Z.J. Aortobronchial fistula as a complication of surgery for correction of congenital aortic anomalies. Radiology 1990;175:347-348.[Abstract/Free Full Text]
5. Caes F., Taeymans Y., Van Nooten G. Aortobronchial fistula: a late complication of coarctation repair by patch aortoplasty. Thorac Cardiovasc Surg 1993;41:80-82.[Medline]
6. Miller J.P., Cammarata S.K. Massive hemoptysis 17 years after repair of aortic coarctation. Chest 1994;105:1249-1250.[Medline]
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8. Demeter S.L., Cordasco E.M. Aortobronchial fistula: keys to successful management. Angiology 1980;31:431-435.

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