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J Thorac Cardiovasc Surg 1997;114:853-856
© 1997 Mosby, Inc.

BRIEF COMMUNICATIONS

RUPTURED MEDIASTINAL BRONCHIAL ARTERY ANEURYSM: A DILEMMA OF DIAGNOSIS AND THERAPEUTIC APPROACH

Geneva, Switzerland

From the Clinic for Cardiovascular Surgery, University Cantonal Hospital of Geneva, Geneva, Switzerland.

Received for publication Feb. 3, 1997; accepted for publication March 17, 1997. Address for reprints: Afksendiyos Kalangos, MD, PhD, Clinic for Cardiovascular Surgery, University Cantonal Hospital of Geneva, 24, rue Micheli-du-Crest, 1211 Geneva, 14, Switzerland.

Bronchial artery aneurysm is a rare entity that is observed in fewer than 1% of all cases of selective bronchial arteriography. ¹ It can be classified as mediastinal, intrapulmonary, or both. The mediastinal localization may manifest various symptoms simulating clinical pictures of aortic dissection or rupture of an aortic aneurysm that could raise some challenging problems. ^2,3 We report a case complicated by life-threatening massive bleeding from a ruptured mediastinal bronchial artery aneurysm mimicking a clinical picture of aortic dissection. The mechanisms of formation, clinical presentation, and therapeutic management of mediastinal bronchial artery aneurysms are discussed with reference to the 13 previously reported cases in the literature (Table I). ^1-11

A 50-year-old man was brought to a local hospital because of the sudden onset of severe back pain radiating to the abdomen during a mountain excursion. Blood pressure was 95/60 mm Hg, pulse rate 110 beats/min, and respiratory rate 18 breaths/min. The breath sounds were decreased over the left hemithorax. The chest x-ray film revealed a left hemothorax with no evidence of mediastinal widening or suspicious images on the pulmonary parenchyma. Chest computed tomographic (CT) scan confirmed the left hemothorax and excluded aortic dissection. However, a suspicious vascular mass in the midportion of the mediastinum, ventral to the descending aorta, was visualized, but its nature could not be identified with certainty (Fig. 1). On referral to our institution, he abruptly went into hypovolemic shock and required massive blood and fluid transfusions. The patient was immediately taken to the operating room. Partial cardiopulmonary bypass was instituted with inflow into the right femoral artery and venous return to the pump from a long cannula placed into the right atrium through the right femoral vein. A perioperative transesophageal echocardiogram excluded the presence of aortic dissection or aortic aneurysm. A bilateral transsternal anterolateral thoracotomy revealed a large blood effusion in the left extrapleural space and 3000 ml of clotted and unclotted blood in the left pleural cavity. A subadventitial hematoma was found along the descending aorta extending toward the aortic arch and the middle mediastinum. The pericardium was opened to determine whether the bleeding had originated in the ascending aorta or the aortic arch, but the appearance and diameter of these two aortic segments were normal. Dissection of the descending aortic adventitia revealed an active bleeding site from a juxtaaortic structure. Further dissection of the middle mediastinum showed that the bleeding was coming from a mediastinal bronchial artery aneurysm, 15 mm in diameter, with a ventral origin from the descending aorta (Fig. 2). It was ligated proximally and distally and the aneurysmal sac was compressed by large vascular clips after biopsy. The postoperative course was uneventful. Histopathologic examination of the specimen revealed no evidence of atherosclerotic or inflammatory changes but did show signs suggestive of medial degeneration characterized by broken elastic laminae.

View larger version (132K): [in this window] [in a new window]   Fig. 1. Chest CT scan showing an abnormal vascular structure ventral to the descending aorta (arrow). Note the presence of the large left hemothorax and the absence of dissection in the descending aorta.

View larger version (124K): [in this window] [in a new window]   Fig. 2. Operative view of the ruptured bronchial artery aneurysm (arrow) at the ventral proximity of the descending aorta. Note the bleeding jet stream from the bronchial artery aneurysm (between arrowheads) toward the sucker.

Bronchial artery aneurysms are reported to occur as isolated or associated mediastinal and intrapulmonary anomalies. They are usually found in cases of bronchiectasis or recurrent bronchopulmonary inflammation. However, in some cases, atherosclerosis, ^3,9 infection, ⁶ trauma, ¹¹ and Rendu-Osler-Weber syndrome ⁴ have been demonstrated to predispose bronchial arteries to aneurysmal dilatation. For the few cases in which the cause has not been elucidated in detail, a proposed hypothesis is that increased bronchial arterial flow to the lungs owing to various factors could be a possible mechanism of bronchial artery dilatation and subsequent aneurysm formation. The formation of our patient's bronchial artery aneurysm raises doubts in favor of medial degenerative changes or the aforementioned hypothesis of increased bronchial artery flow to the lungs, because no associated bronchopulmonary, atherosclerotic, or mycotic origin was demonstrated. Bronchial artery aneurysms can stay asymptomatic until rupture. Although little is known about the process that leads the aneurysm to rupture, the various sizes of ruptured mediastinal bronchial artery aneurysms in reported cases have revealed that diameter is not an incremental risk factor for rupture. Rupture into the pulmonary parenchyma mainly presents as hemoptysis, whereas a rupture into the mediastinum, in the pleural space, ^2-4 bronchus, ^6,8,10,11 or esophagus, ⁹ may simulate symptoms of aortic dissection or aortic rupture with subsequent shock. The severe tearing pain in the central portion of the chest radiating into the back and epigastrium is due to blood stripping from the mediastinum, containing the ruptured extraaortic vessel into the subadventitial aortic and extrapleural areas. When adventitial or pleural barriers are broken, the clinical picture is that of a ruptured thoracic aneurysm characterized by potentially life-threatening hypovolemic shock. In these instances in which time does not allow for diagnostic tools, exploratory thoracotomy could incidentally identify a ruptured mediastinal bronchial artery aneurysm as the bleeding site and allow treatment. The use of a partial femoro-femoral bypass may be life-saving in patients who are in hemodynamically unstable condition. Moreover, in patients who are in hemodynamically stable condition, femoro-femoral bypass could also be a safe approach, better assuring the surgical exploration of ruptured bronchial artery aneurysm and thereby avoiding problems of uncontrollable bleeding. Mediastinal bronchial artery aneurysm can also be incidentally identified on chest CT scans ^8,9 or exceptionally on conventional chest x-ray films ¹ during investigations of bronchiectasis or bronchopulmonary diseases. In our patient, a contrast chest CT scan performed in a local hospital demonstrated the presence of a suspicious mediastinal mass near the descending aorta, but the variety of vascular structures in the mediastinum that could interfere with interpretation did not permit the diagnosis of ruptured bronchial artery aneurysm to be confirmed with certainty. Although contrast CT scan reveals a high index of suspicion for mediastinal or intrapulmonary bronchial artery aneurysm, ¹⁰ definite diagnosis is confirmed by selective bronchial artery angiography, which additionally enables therapeutic embolization with either balloons or steel coils. ⁴ Transcatheter embolization of a bronchial artery may be successful in obliterating the blood flow to the aneurysm and stopping bleeding in hemodynamically stable patients with no evidence of life-threatening bleeding. Embolization succeeded in excluding the aneurysm in three cases of mediastinal bronchial artery aneurysm. ^4,8,11 However, embolization of the aneurysm in which only a short segment exists between the aneurysm and aorta could cause iatrogenic complications and render the procedure inefficient. ¹⁰ In cases of mediastinal bronchial artery aneurysm manifested by hemoptysis, lobectomy seems to be the standard surgical technique, with four of the five reported cases being managed in this manner. In one case, lobectomy was performed after bleeding was first controlled by transcatheter embolization. ⁸

We have stressed that incidentally detected mediastinal bronchial artery aneurysms of any cause cannot be assumed to be stable, and in the light of the collected case histories wherein spontaneous rupture led to life-threatening bleeding, intervention should be favored, regardless of the presence or absence of symptoms. In addition, because of the variety of clinical pictures of ruptured bronchial artery aneurysms mimicking other vascular entities and the various mediastinal vessels causing confusion, the identification of the vascular nature of a mediastinal mass in hemodynamically stable patients should be mandatory to better guide the type of therapeutic approach.

References

1. Fujita J, Akashi K, Kunikane H, et al. A case of bronchial artery aneurysm demonstrating a mass shadow on chest x-ray film. Nippon Kyobu Shikkan Gakkai Zasshi 1991;29:1591-5.
   
2. Hall RJC, Miller GAH, Kerr IH. Ruptured bronchial artery aneurysm mimicking aortic dissection. Br Heart J 1977;39:909-10. [Abstract/Free Full Text]
   
3. Abet D, Pietri J. Rupture d'anévrysme d'une artère bronchique, sur séquestration pulmonaire, simulant une dissection aortique. J Chir 1981;118:743-6.
   
4. Ishizaki N, Shimokawa S, Tanaka K, et al. Ruptured bronchial artery aneurysm associated with pleural telangiectasis and tortuous portal obstruction: report of a case. Jpn J Surg 1995;25:852-4.
   
5. Hirano N, Maeda H. Two cases of bronchial artery aneurysm. Nippon Igaku Hoshasen Gakkai Zasshi (J Jpn Radiol Soc)1976;36:82.
   
6. Chantepie A, Robert M, Pelletier J, et al. Anévrysme mycotique d'une artère bronchique. Chir Pediatr 1980;21:407-10. [Medline]
   
7. Watanabe S, Matayoshi Y, Takeshita H, et al. Two cases of bronchial artery aneurysm. Hirosaki Med J 1981;33:512-3.
   
8. Osada H, Kawada T, Ashida H, Sodemoto Y, Noguchi T. Bronchial artery aneurysm. Ann Thorac Surg 1986;41:440-2. [Abstract]
   
9. Shaer AH, Bashist B. Computed tomography of bronchial artery aneurysm with erosion into the esophagus. J Comput Assist Tomogr 1989;13:1069-71. [Medline]
   
10. Remy-Jardin M, Remy J, Ramon P, Fellous G. Mediastinal bronchial artery aneurysm: dynamic computed tomography appearance. Cardiovasc Intervent Radiol 1991;14:118-210. [Medline]
    
11. Cearlock JR, Fontaine AB, Urbaneja A Spigos DG. Endovascular treatment of a posttraumatic bronchial artery pseudoaneurysm. J Vasc Interv Radiol 1995;6:495-6.[Medline]
    

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