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Ann Thorac Surg 2006;82:187-190
© 2006 The Society of Thoracic Surgeons


Original article: Cardiovascular

Surgical Treatment of Aberrant Right Subclavian Artery (Arteria Lusoria) Aneurysm Using Three Different Methods

Hiroyuki Kamiya, MD a , b , * , Karsten Knobloch, MD a , Joachim Lotz, MD a , Antje Bog, MD a , Artur Lichtenberg, MD a , Christian Hagl, MD a , Klaus Kallenbach, MD a , Axel Haverich, MD a , Matthias Karck, MD a

a Division of Thoracic and Cardiovascular Surgery, Hannover Medical School, Hannover, Germany
b Division of Radiology, Hannover Medical School, Hannover, Germany

Accepted for publication February 22, 2006.

* Address correspondence to Dr Kamiya, Division of Thoracic and Cardiovascular Surgery, Hannover Medical School, Carl-Neuberg-Strasse 1, 30625 Hannover, Germany (Email: hkamiya88{at}yahoo.co.jp).


    Abstract
 Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 
BACKGROUND: Here we report our surgical experiences with aberrant right subclavian artery (ARSA) aneurysm in 8 patients.

METHODS: Eight patients underwent surgical treatment for ARSA aneurysm between March 1994 and June 2005. The age of these patients ranged from 20 to 75 years. The mean size of the ARSA aneurysm was 3.3 cm, ranging from 2 to 5 cm. The ARSA aneurysm was completely resected through a left posterolateral thoracotomy after reconstruction of the right subclavian artery through the supraclavicular approach in 4 patients (group 1). The ARSA aneurysm was excluded through a left posterolateral thoracotomy without revascularization of the right subclavian artery in 2 patients (group 2). The distal site of the ARSA aneurysm was closed followed by revascularization through a median sternotomy, and the ARSA aneurysm was left as a blind sack in 2 patients (group 3).

RESULTS: None of the patients in group 1 or 3 had any postoperative complications. In group 2, 1 had a steal syndrome caused by the exclusion of the ARSA aneurysm, and the other died of sepsis 2 months after the operation.

CONCLUSIONS: Complete anatomical repair of the ARSA aneurysm could be performed through the combination of the supraclavicular approach and the left posterolateral thoracotomy, with excellent results. Exclusion of the ARSA aneurysm without revascularization resulted in a suboptimal outcome. Surgical results of simple closure of the ARSA followed by revascularization were uneventful, but the ARSA aneurysm was left as a blind sack.


    Introduction
 Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 
The aberrant right subclavian artery (ARSA), also called arteria lusoria, is a rare entity of a congenital aortic arch anomaly, with a reported prevalence as high as 2% [1, 2]. Most patients with ARSA are asymptomatic, but aneurysms in this location can cause serious complications, such as distal embolization, rupture, and compression of neighboring structures [3]; therefore, its surgical treatment is crucial. However, surgical strategies are yet unclear because of the rareness of the ARSA aneurysm, with a limited number of reported cases. Here, we report our surgical experiences with aneurysm of ARSA in 8 patients.


    Patients and Methods
 Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 
Eight patients underwent surgical treatment for ARSA aneurysm between March 1994 and June 2005. The age of these patients ranged from 20 to 75 years, with an average age of 55.8 years. There were 4 men and 4 women. Those patients were retrospectively examined, and the Review Board of our institute approved this retrospective study.

Diagnoses of the patients are shown in Table 1. Only 1 patient (patient 1) had dyspnea as a postoperative symptom, which appeared to be caused by an ARSA aneurysm. Two patients (patients 3 and 5) with chronic aortic dissection type B and patient 2 with descending aortic aneurysm 20 years after the correction of the aortic coactation were asymptomatic. Two patients with subacute aortic dissection type B suffered from back pain, patient 7 had dyspnea due to aortic regurgitation, and patient 8 had dyspnea and fever due to prosthesis-valve endocarditis. Elective operation was performed in 5 patients, and the others underwent emergency operations (patients 4, 6, and 8). Surgical treatment only to resect the ARSA aneurysm was indicated in 1 patient (patient 1), and the ARSA was treated as an additional lesion in the other patients. The mean size of the ARSA aneurysm was 3.3 cm, ranging from 2 to 5 cm.


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Table 1. Patient Characteristics and Surgical Intervention
 
Surgical procedures are listed also in Table 1. The ARSA aneurysm was completely resected using two surgical approaches in 4 patients (group 1, patients 1 through 4; Fig 1A). A carotid-subclavian bypass or a translocation of the right subclavian artery onto the right common carotid artery with closure of the distal ARSA was at first performed in those patients with a supraclavicular approach in the supine position. For this anastomosis, side-clamping of the right common carotid artery was performed. Then, the ARSA aneurysm was resected through a left posterolateral thoracotomy, with resection and replacement of the descending aorta using cardiopulmonary bypass. Three patients underwent two-staged operations with an interval ranging from 1 week to 4 months considering the stable status of the patients, and 1 patient underwent a one-staged operation with repositioning of the patient as an emergency case.


Figure 1
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Fig 1. Schematic presentation of the three differential surgical approaches for the aberrant right subclavian artery (ARSA) aneurysm. (Left) Complete resection of the ARSA aneurysm. (Center) Exclusion of the ARSA aneurysm. (Right) Closure of the ARSA (the ARSA aneurysm was left as a blind sack).

 
The ARSA aneurysm was excluded through a left posterolateral thoracotomy in 2 patients (group 2, patients 5 and 6; Fig 1B). They had a descending aortic aneurysm and aneurysmatic enlargement of the ARSA. Resection and replacement of the descending aorta was performed through a posterolateral thoracotomy using cardiopulmonary bypass, and the proximal site of the ARSA aneurysm was closed at the same time. The right subclavian artery was not revascularized in those 2 patients.

The distal site of the ARSA aneurysm was closed through a median sternotomy, and the right subclavian artery and the ARSA aneurysm was left as a blind sack in 2 patients (group 3, patients 7 and 8; Fig 1C). They received replacement of the ascending aorta using cardiopulmonary bypass, and the right subclavian artery was revascularized with an aorto–right subclavian bypass (patient 7) or a translocation of the right subclavian bypass onto the right common carotid artery (patient 8). In patient 7, the aneurysm was relatively small (3 cm), but this procedure was done as a preparation for the second operation at some time because the patient was young. The resection of the ARSA aneurysm was attempted in both cases, but the proximal site of the ARSA aneurysm could not be controlled through a median thoracotomy because the position of the ARSA aneurysm was too deep (Fig 2).


Figure 2
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Fig 2. Computed tomography findings of patient 3 after the carotid-right subclavian bypass with closure of the aberrant right subclavian artery (ARSA) and before the replacement of the descending aorta. (Left) The ARSA aneurysm, the right subclavian artery was anastomosed to the right common carotid artery. (Right) Relationship between the ARSA aneurysm and the trachea.

 

    Results
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 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 
All patients survived the operations and were discharged from the hospital. None of the patients in groups 1 or 3 had any postoperative complications.

Both patients in group 2 had complicated postoperative courses. Patient 5, who received replacement of the descending aorta and exclusion of an ARSA aneurysm, suffered from dizziness directly after the operation, and the symptom was worsened when the right arm was moved. The angiography revealed a significant blood flow through the right vertebral artery to the right subclavian artery, and it was diagnosed as a steal syndrome caused by the exclusion of the ARSA aneurysm. The patient underwent a right carotidosubclavian bypass 3 months after the initial operation in another hospital. Patient 6, who received the same procedure as patient 5 owing to acute aortic dissection type B and an accompanying ARSA aneurysm, suffered from postoperative respiratory failure and presented with delayed awakening. A cerebral computed tomography scan was done on the eighth postoperative day but no cerebral insult was detected. He was transferred to another hospital for further intensive care on the 13th postoperative day, and died of sepsis 2 months after the operation.


    Comment
 Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 
Aberrant right subclavian artery aneurysm is an extremely rare entity. In our institute, approximately 2,000 cardiac operations are performed each year, but there have been only 8 operated cases of this aneurysm in the last 12 years. Because of the rareness of this disease, the mortality and morbidity rates of its surgical treatment are still unclear. Some case reports on the surgical treatment of ARSA aneurysm have been published, but it is very difficult to ascertain the mortality and morbidity associated with this operation from those case reports because it is likely that only successful cases have been published.

The report by Kieffer and colleagues [4] published in 1994 is the only available data source of a large series of the surgical treatment of ARSA aneurysm. Seventeen patients with an aneurysm of the aberrant subclavian artery were presented in their report. Thirteen patients had a left-sided aortic arch and an ARSA aneurysm, and the other 4 patients had a right-sided aortic arch and an aberrant left subclavian artery aneurysm. Of these 17 patients, 4 patients died after the operation (24% mortality): 2 of multiorgan failure, 1 of heart failure, and 1 of esophageal rupture. The mortality of the surgical treatment of the ARSA aneurysm cannot be discerned from the report, because their report includes cases of an aberrant left subclavian artery aneurysm associated with a right-sided aortic arch. Nevertheless, the risk of the surgical treatment of ARSA aneurysm can be roughly estimated from their report. In our present series, 1 patient died 2 months after the surgery (mortality: 12%) and 1 suffered from postoperative subclavian steal syndrome (morbidity: 12%). These results appear to be acceptable, and it is considered that surgical results of the treatment of ARSA aneurysms have been improved compared with the times of those reported by Kieffer and associates [4], owing to the development of surgical and perioperative management in the last decade.

Because of the anatomical position of ARSA aneurysms, it is very difficult to treat them completely only by one approach (supraclavicular approach, median sternotomy, or left thoracotomy). In this series, patients in group 1 underwent complete anatomical repair of the ARSA aneurysm with resection of the aneurysm and reconstruction of the right subclavian artery through a supraclavicular approach in the supine position and a left posterolateral thoracotomy, and those surgical results were excellent without complications. Thus, according to our experience, this method using the supraclavicular approach and a left thoracotomy enables secure and complete repair of ARSA aneurysm. On the other hand, it may be difficult to apply it to an emergency situation, although 1 patient with sealed rupture of the descending aorta and an accompaning ARSA aneurysm underwent this method in our series.

The surgical results of the 2 patients in group 2 were suboptimal. They underwent replacement of the descending aorta and exclusion of an ARSA aneurysm through a left thoracotomy. In this approach, revascularization is impossible. One patient (patient 6) died of sepsis, although it seemed that nonrevascularization of the right subclavian artery played no roll in his severe postoperative course. However, the other (patient 5) suffered from subclavian steal syndrome, and this complication was directly associated with nonrevascularization. In this patient, the possibility of postoperative subclavian steal syndrome was underestimated, and therefore revascularization of the right subclavian artery was not done. The cause of the occurrence of the subclavian steal syndrome after a simple closure of the right subclavian artery is not clear; however, it might be advisable to revascularize the right subclavian artery if possible, especially since the revascularization of the right subclavian artery with grafting or translocation is relatively easy to perform, normally within 1 hour.

The postoperative courses of the patients in group 3 were uneventful. The right subclavian artery was revascularized, but the ARSA aneurysm was left as a blind sack. In these patients, the main lesion that required surgical intervention was the ascending aorta, and resection of the accompaning ARSA aneurysm was not possible through a median sternotomy in our series. For such patients, there are two surgical options: a one-stage operation using deep hypothermic circulatory arrest, or a two-stage surgical and endovascular treatment. Kokotsakis and colleagues [5] and Kono and associates [6] presented complete surgical repair of ARSA aneurysm using deep hypothermic circulatory arrest; and Attmann and coworkers [7] and Corral and colleagues [8] reported treatment of ARSA aneurysm with endovascular exclusion and adjunctive surgical bypass. Our patients in group 3, especially patient 8, were too ill to tolerate deep hypothermic circulatory arrest, and therefore revascularization was performed as a preparation for the second operation, optimally using the endovascular method.

In conclusion, ARSA aneurysm was treated in 8 patients. Complete anatomical repair of the ARSA aneurysm with resection of the aneurysm and reconstruction of the right subclavian artery could be performed through the supraclavicular approach in the supine position and a left posterolateral thoracotomy, and those surgical results were excellent without complications. This approach is therefore considered to be the optimal strategy for treatment of ARSA aneurysm when endovascular treatment with revascularization of the right subclavian artery is not possible. On the other hand, exclusion of the ARSA aneurysm without revascularization of the right subclavian artery resulted in a suboptimal outcome. The postoperative courses of patients who received cardiac operation, ligation of the distal site of the ARSA aneurysm, and revascularization of the right subclavian artery were uneventful, but the ARSA aneurysm was left as a blind sack.


    References
 Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 

  1. Boening A, Dresler C, Haverich A, Cremer J. Two-stage repair of a combined aneurysm of the descending aorta and the aberrant right subclavian artery Eur J Cardiothorac Surg 1999;16:246-248.[Abstract/Free Full Text]
  2. Freed K, Low VH. The aberrant subclavian artery AJR Am J Roentgenol 1997;168:481-484.[Free Full Text]
  3. Austin EH, Wolfe WG. Aneurysm of aberrant subclavian artery with a review of the literature J Vasc Surg 1985;2:571-577.[Medline]
  4. Kieffer E, Bahnini A, Koskas F. Aberrant subclavian arterysurgical treatment in thirty-three adult patients. J Vasc Surg 1994;19:100-109.[Medline]
  5. Kokotsakis JN, Lazopoulos GL, Lioulias AG, Kaskarelis JS, Romana CN, Lolas CT. Surgical treatment for aneurysm at the origin of an aberrant right subclavian artery Ann Vasc Surg 2003;17:315-319.[Medline]
  6. Kono T, Kitahara H, Watanabe T, Takano T, Sakaguchi M, Amano J. Successful repair of aberrant right subclavian artery aneurysm combined with bicuspid aortic valve through a median sternotomy Ann Thorac Surg 2004;77:2196-2197.[Abstract/Free Full Text]
  7. Attmann T, Brandt M, Muller-Hulsbeck S, Cremer J. Two-stage surgical and endovascular treatment of an aneurysmal aberrant right subclavian (Lusoria) artery Eur J Cardiothorac Surg 2005;27:1125-1127.[Abstract/Free Full Text]
  8. Corral JS, Zuniga CG, Sanchez JB, Guaita JO, Basail AM, Gimeno CC. Treatment of aberrant right subclavian artery aneurysm with endovascular exclusion and adjunctive surgical bypass J Vasc Interv Radiol 2003;14:789-792.[Medline]



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