Ann Thorac Surg 2006;82:e35-e36
© 2006 The Society of Thoracic Surgeons
Case Reports
Tuberculous Hilar Lymph Nodes Leading to Tracheopulmonary Artery Fistula and Pseudoaneurysm of Pulmonary Artery
Saulat H. Fatimi, MD, FACSa,
Muhammad A. Javed, MDa,*,
Usman Ahmad, MBBSa,
Buland I. Siddiqi, MBBSa,
Nawal Salahuddin, MD, FACCPb
a Department of Surgery, Division of Cardiothoracic Surgery, Aga Khan University, Karachi, Pakistan
b Department of Medicine, Division of Pulmonology, Aga Khan University, Karachi, Pakistan
Accepted for publication June 28, 2006.
* Address correspondence to Dr Javed, Department of Surgery, Division of Cardiothoracic Surgery, Aga Khan University Hospital, Stadium Rd, Karachi 74800 Pakistan. (Email: ahsanjaved{at}yahoo.com).
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Abstract
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Tracheopulmonary artery fistula is an extremely rare condition. We report the case of an 80-year-old man who presented with massive hemoptysis. He was found to have tuberculous mediastinal lymph nodes that had eroded into the pulmonary artery and anterior tracheal wall, leading to a pseudoaneurysm of the right pulmonary artery and a tracheopulmonary artery fistula.
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Introduction
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Fistulas between the systemic arteries and the trachea are usually a result of aneurysm erosion or tracheal trauma due to intubation or erosion of the tracheostomy cuff and have been well described. A fistulous communication between the trachea and the pulmonary artery is quite unusual, however, and only one previous report has been described [1]. A pseudoaneurysm of the pulmonary artery is also a rare entity and has been associated with tuberculosis, syphilis, infective endocarditis, congenital heart disease, trauma, thromboembolism, intravenous drug abuse, and pulmonary artery catheterization [2, 3]. We present a unique case of massive hemoptysis secondary to hilar lymph node tuberculosis that had led to the formation of a tracheopulmonary artery fistula and a pulmonary artery pseudoaneurysm.
An 80-year-old man presented after two episodes of massive hemoptysis, each of about 750 mL. His only symptom was a 6-month history of shortness of breath. On examination he was tachycardiac, tachypneic, and had bilateral extensive coarse crepitations and expiratory rhonchi.
The result of a chest roentgenogram was unremarkable. Computed tomography of the chest (Fig 1) with contrast showed an enhancing soft-tissue mass in the right hilar region extending proximally for 2 cm along the right side of the trachea. Extraluminal air was seen around the trachea. A soft-tissue mass measuring 3 x 2 x 1.5 cm was identified in the apical segment of the right upper lobe. Bronchoscopy showed mucosal swelling and irregularity in the lower 2 cm of the trachea, with a fistulous opening in the anterior wall. The carina was widened along with a grossly abnormal left main bronchus with swollen and irregular mucosa and narrowing of the lumen. A pulmonary angiogram revealed a moderately sized pseudoaneurysm arising from the anterior trunk of the right main pulmonary artery (Fig 2).
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Fig 1. Dynamic computed tomography shows diffuse soft-tissue mass infiltrating around the right pulmonary artery in the right hilar region (white arrow). Pneumomediastinum can also be identified (black arrow).
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Fig 2. Right pulmonary angiogram shows pseudoaneurysm (arrow) arising from anterior trunk of pulmonary artery.
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The patient underwent an emergent operation. After establishing cardiopulmonary bypass, the pulmonary artery was dissected from the aorta and dissection was extended up to the right lung hilum. The superior vena cava was dissected all around and separated from the right pulmonary artery.
A fistula was identified involving the trachea and the right pulmonary artery, near the origin of the anterior trunk. Several matted tracheobronchial/subcarinal lymph nodes were identified in this area. The operative findings suggested that the fistula was formed due to erosion of the matted lymph nodes into pulmonary artery and into the anterior wall of the lower trachea. The fistulous communication was confirmed by intraoperative bronchoscopy and was localized to the anterior tracheal wall, located 1.5 cm proximal to the carina.
The fistulous communication was exposed after a longitudinal pulmonary arteriotomy and closed from inside the pulmonary artery with a pericardial patch. The lymph nodes adjacent to the right pulmonary artery and the tract of the fistula were exposed and excised. There was intense inflammation at the tracheal end of the fistulous tract; therefore, this opening was left unclosed to heal spontaneously with fibrosis. Several lymph nodes were adhering strongly to the trachea and were not excised. The patient was weaned off cardiopulmonary bypass.
Histopathologic analysis of the lymph nodes revealed caseating granulomas consistent with lymph node tuberculosis. The patient was started on appropriate antituberculous treatment and discharged from the surgery service in 2 weeks.
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Comment
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Pulmonary tuberculosis with its chronic complications is the most common cause of hemoptysis in the developing world [4]. Here, we report massive hemoptysis secondary to the formation of a tracheopulmonary artery fistula caused by the erosion of a tuberculous lymph node. A Medline literature search with appropriate key words revealed only one case of tracheopulmonary artery fistula [1], which was secondary to a Palmaz stent (Cordis, Miami Lakes, Fla) insertion for treatment of persistent tracheal stenosis. In that case, multiple attempts to expand the stent balloon led to formation of tracheopulmonary artery fistula. That patient was managed surgically. Several cases of pulmonary artery-bronchial fistulas have been reported [5–7].
A surgical procedure is the treatment of choice for such arteriobronchial and arteriotracheal fistulas. Abruzzini [8] first described the use of median sternotomy for approaching main bronchus fistulas in 1961, and since then, several centers have reported the successful use of this approach. Median sternotomy was also the preferred approach in our patient because it gave the best access to the proximal right pulmonary artery and trachea and allowed us to go on cardiopulmonary bypass at the same time. The right pulmonary artery was dissected from the surrounding structures to assess the repair. The pericardial patch was judged to provide sufficient strength to the repaired fistula and eliminated the need for external repair and support. The procedure needs to be tailored according to the extent of disease and the intraoperative findings.
Our patient also had a pseudoaneurysm of the pulmonary artery, and this was most likely due to erosion of tuberculous lymph nodes into the surrounding structures. The incidence of tuberculosis is increasing worldwide. It is therefore important to be aware of the atypical presentations of tuberculosis and their appropriate management. Abnormal fistulous communications with blood vessels should be considered when evaluating massive hemoptysis.
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References
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- Miyamoto T, Ishida R, Noma M, Chikada M, Sekiguchi A. Successful surgical management of a tracheopulmonary artery fistula caused by an intratracheal expandable metal stent Jpn J Thorac Cardiovasc Surg 2001;49:632-634.[Medline]
- Morgan J, Morgan A, Addis B, Bradley G, Spiro S. Fatal haemorrhage from mycotic aneurysms of the pulmonary artery Thorax 1986;41:70-74.[Free Full Text]
- Roush K, Scala-Barnett D, Donabedian H, Freimer E. Rupture of pulmonary artery mycotic aneurysm associated with candidal endocarditis Am J Med 1988;84:142-144.[Medline]
- Syabbalo N. Hemoptysis: the third world perspective Chest 1991;99:1316-1317.
- Shilyansky J, Wilson W, Temeck BK, Pass HI. Pulmonary artery-bronchial fistula during lymphoma treatment J Thorac Cardiovasc Surg 1994;108:790-791.[Free Full Text]
- Kessler R, Massard G, Warter A, Wihlm JM, Weitzenblum E. Bronchial-pulmonary artery fistula after unilateral lung transplantation: a case report J Heart Lung Transplant 1997;16:674-677.[Medline]
- Guibaud JP, Laborde MN, Dubrez J, et al. Surgical repair of an aneurysm of the innominate artery with fistulization into the trachea Ann Vasc Surg 2001;15:412-414.[Medline]
- Abruzzini P. Trattamen to delle fistole del bronco principale consecutive a pneumonectomia per tubercolosi Chir Toracica 1961;14:165-171.
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Abstract
Introduction
