Ann Thorac Surg 2008;86:306-308. doi:10.1016/j.athoracsur.2008.01.033
© 2008 The Society of Thoracic Surgeons
Case Reports
Bronchial Artery Aneurysm Refractory to Transcatheter Embolization
Jules Lin, MD,
Douglas E. Wood, MD*
Division of Cardiothoracic Surgery, University of Washington, Seattle, Washington
Accepted for publication January 15, 2008.
* Address correspondence to Dr Wood, University of Washington, Division of Cardiothoracic Surgery, 1959 NE Pacific St, Box 356310, Seattle, WA 98195-6310 (Email: dewood{at}u.washington.edu).
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Abstract
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Bronchial artery aneurysms occur rarely, but they are potentially life threatening and require treatment to avoid rupture and other complications. A 37-year-old man, who was otherwise healthy, presented with chest discomfort and was found to have a large right bronchial artery aneurysm. Despite two attempts at embolization, the aneurysm continued to have persistent flow. Aneurysmectomy with ligation of multiple collateral vessels was successfully performed through a standard posterolateral thoracotomy. Recovery was uneventful, and the patient was discharged on postoperative day 2.
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Introduction
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Bronchial artery aneurysms are rare, and their cause is unclear. However, they have been associated with chronic pulmonary inflammation, bronchiectasis, pulmonary agenesis, and vascular abnormalities [1]. When symptomatic, they can cause hemoptysis and chest pain. The presentation depends on the location and size of the aneurysm and can result from free rupture, compression, or fistula to adjacent structures. They are potentially life threatening due to massive hemoptysis or rupture. We present a patient with persistent flow in a right bronchial artery aneurysm despite two attempts at transcatheter embolization.
The patient is an otherwise healthy 37-year-old man who initially presented with chest discomfort. He denied any history of hemoptysis or thoracic trauma. Chest roentgenograms (Fig 1A) revealed a right hilar mass. Further work-up included a chest computed tomographic scan that showed a 4 x 4 cm right bronchial artery aneurysm (Figs 1B–1D). A selective right bronchial arteriogram confirmed the diagnosis, as well as significant collaterals from the right internal mammary artery (Fig 2). The patient then underwent two attempts for embolization at an outside institution. However, due to extensive collaterals, there was persistent flow in the aneurysm. To prevent potential complications, including rupture and embolization to the systemic circulation, it was recommended that he undergo surgical resection.
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Fig 1. (A) Chest roentgenogram obtained for evaluation of chest discomfort shows a right hilar mass. (B) Contrast-enhanced computed tomographic scan shows a 4 cm x 4 cm right bronchial artery aneurysm on mediastinal windows, as well as (C) coronal and (D) sagittal views.
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Fig 2. (A) A right bronchial artery aneurysm is confirmed on selective right bronchial arteriogram. (B) Multiple collateral vessels are seen including a prominent collateral originating from the right internal mammary artery.
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A serratus-sparing right posterolateral thoracotomy was performed. A well-demarcated 4-cm aneurysm was identified at the junction of the major and minor fissures (Fig 3A). Feeding vessels from the right internal mammary and bronchial arteries were identified as they coursed through the mediastinum. After mobilizing the pulmonary ligament, the remainder of the bronchial arterial blood supply was divided serially from the superior aspect of the inferior pulmonary vein up to the carina following the medial aspect of the bronchus intermedius and mainstem bronchus. Several large collateral vessels were identified and ligated. After careful dissection from adjacent lung parenchyma, the underlying airway, and the pulmonary artery, the aneurysm was excised (Figs 3B and 3C). Postoperatively, the patient's recovery was uneventful, and he was discharged home on postoperative day 2. Follow-up at 2 and 6 weeks after surgery demonstrated a normal chest x-ray film and complete recovery from the thoracotomy without complications. Pathology showed attenuation of the elastic fibers with atherosclerotic changes consistent with an aneurysm.
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Fig 3. (A) The bronchial artery aneurysm, located at the junction of the major and minor fissures, has been dissected free from adjacent lung parenchyma. The aneurysm was then freed from the underlying airway and pulmonary artery. (B) Multiple collateral vessels were ligated. (C) The excised specimen is shown.
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Comment
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The cause of bronchial artery aneurysms is unknown, but aneurysm formation is believed to be associated with increased bronchial artery blood flow and weakening of the vessel wall. They can be congenital with pulmonary sequestration or pulmonary artery agenesis. Other conditions associated with bronchial artery aneurysms include inflammatory conditions, such as bronchiectasis, silicosis, and infections such as tuberculosis. Bronchial artery aneurysms can also be secondary to trauma and vascular degenerative diseases [2]. The patient presented here is interesting because he was young and otherwise healthy without these comorbidities.
The location of the bronchial artery aneurysms can be mediastinal or intrapulmonary. Mediastinal bronchial artery aneurysms may cause symptoms due to compression or rupture into adjacent structures. Intrapulmonary bronchial artery aneurysms present with hemoptysis, which is the most frequent symptom. When patients have chest pain, the presentation can be confused with an acute aortic dissection [3]. Superior vena cava syndrome from external compression [4] and hematemesis from erosion into the esophagus have also been reported [5].
Bronchial artery aneurysms are potentially life threatening. Diameter alone does not seem to be an independent risk factor [1], and once the diagnosis is confirmed, the aneurysm should either be embolized using a transcatheter approach or resected surgically regardless of symptoms. During the past few years, transcatheter embolization has become more common. Care must be taken to identify and coil all collateral vessels, both afferent and efferent. The bronchial arteries usually originate directly from the aorta, but the right bronchial artery can also arise from the first or second intercostal or the right internal mammary artery. Collateral branches often lead to the esophagus and hilar lymph nodes. Follow-up imaging is required after embolization, as there have been reports of recurrent hemoptysis and persistent perfusion of the aneurysm due to revascularization through collateral vessels [2]. Embolization can also be difficult when the segment between the aorta and the bronchial artery aneurysm is short. Operative resection is performed when the transcatheter approach is not feasible or unsuccessful and provides definitive treatment. The two approaches can also be complementary [6].
Bronchial artery aneurysms are potentially life threatening and should be treated when diagnosed regardless of symptoms. They can be approached endovascularly when feasible using transcatheter embolization, although flow in the aneurysm may be persistent unless all afferent and efferent collaterals are identified and ablated. In cases in which transcatheter embolization is unsuccessful or not feasible, aneurysmectomy provides a reliable alternative approach.
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References
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- Kalangos A, Khatchatourian G, Panos A, Faidutti B. Ruptured mediastinal bronchial artery aneurysm: a dilemma of diagnosis and therapeutic approach J Thorac Cardiovasc Surg 1997;114:853-856.[Free Full Text]
- Sancho C, Dominguez J, Escalante E, Hernandez E, Cairols M, Martinez X. Embolization of an anomalous bronchial artery aneurysm in a patient with agenesis of the left pulmonary artery J Vasc Interv Radiol 1999;10:1122-1126.[Medline]
- Tanaka K, Ihaya A, Horiuci T, et al. Giant mediastinal bronchial artery aneurysm mimicking benign esophageal tumor: a case report and review of 26 cases from literature J Vasc Surg 2003;38:1125-1129.[Medline]
- Hoffman V, Ysebaert DeSchepper A, Colpaert C, Jorens P. Acute superior vena cava obstruction after rupture of a bronchial artery aneurysm Chest 1996;110:1356-1358.[Medline]
- Shaer AH, Bashist B. Computed tomography of bronchial artery aneurysm with erosion into the esophagus J Comput Assist Tomogr 1989;13:1069-1071.[Medline]
- Nakajima H, Haneda T, Kambayashi M, et al. Two giant bronchial aneurysms: effect of preoperative embolisation. case report. Eur J Surg 1995;161:855-856.[Medline]
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Abstract
Introduction
