Ann Thorac Cardiovasc Surg 2015; 21: 564–566
Case Report
Online June 5, 2015 doi: 10.5761/atcs.cr.15-00054
A Surgical Case of Bronchial Artery Aneurysm Directory Connecting with Pulmonary Artery Akihiko Kitami, MD,1 Fumitoshi Sano, MD,1 Shoko Hayashi, MD,1 Kosuke Suzuki, MD,1 Shugo Uematsu, MD,1 Takashi Suzuki, MD,1 and Noriyuki Saeki, MD2
We present a surgical case of bronchial artery aneurysm (BAA) connecting pulmonary artery accompanied with racemose hemangioma. This is a third surgical case report of BAA directly connecting pulmonary artery in the English literature. A 63-year-old female was found a BAA, 2 cm in diameter, connecting right A4 pulmonary artery. The patient underwent two attempts for embolization. However, due to extensive collaterals, there was persistent flow in the aneurysm. Standard lateral thoracotomy was performed. A BAA was located between A4 and A5 PA. A small branch of A4 PA was separated, and the small vessel connecting to the BAA could be ligated. A5 PA was separated similarly, however BAA was ruptured not to identify the other small vessel connecting to the BAA. After a clamp of the BAA, middle lobe lobectomy was performed. We removed the aneurysm with dilated bronchial artery connecting to the aneurysm. The postoperative course was uneventful. Keywords: bronchial artery aneurysm, connection to the pulmonary artery, racemose hemangioma
Introduction
Case Report
Bronchial artery aneurysm (BAA) is rare, but they are potentially life threatening and require treatment to avoid rupture and other complications. We present a surgical case of bronchial artery aneurysm connecting directly right pulmonary artery. To our knowledge, a few cases of bronchial artery aneurysm connecting pulmonary artery has been reported in Japanese literature,1,2) and only three in English literature.3–5) This is a third surgical case report of bronchial artery aneurysm directly connecting pulmonary artery in the English literature.
A 63-year-old female was found to have an abnormal shadow in a regular checkup chest X-ray film in 2005. She had no symptoms such as bloody sputum, hemoptysis or cardiac failure. Chest computed tomography (CT) scan revealed a round mass adjacent to the pulmonary artery in the right hilum, and bronchial artery dilated like vine; so called racemose hemangioma. Aortography, bronchial arteriography and pulmonary arteriography showed a bronchial artery aneurysm, 2 cm in diameter, connecting right pulmonary artery. The patient underwent two attempts for embolization at another hospital in 2006 and 2007. However, due to extensive collaterals, there was persistent flow in the aneurysm. The patient was referred to our hospital in 2011 for the purpose of surgical resection. Bronchoscopy revealed a submucous elevated lesion in the right middle bronchus (Fig. 1). Three dimensional computed tomography (3-D CT) demonstrated a narrow vessel which is connected aneurysm and right A4 pulmonary artery (Fig. 2). Standard lateral thoracotomy was performed via the fifth. A BAA 20 mm in diameter with thrill was located between
Respiratory Disease Center, Showa University Northern Yokohama Hospital, Yokohama, Kanagawa, Japan 2Department of Thoracic Surgery, Ofuna Chuo Hospital, Kamakura, Kanagawa, Japan 1
Received: February 17, 2015; Accepted: March 20, 2015 Corresponding author: Akihiko Kitami, MD. Respiratory Disease Center, Showa University Northern Yokohama Hospital, 35-1 Chigasaki– Chuo, Tsuzuki-ku, Yokohama, Kanagawa 224-0032, Japan E-mail: [email protected] ©2015 The Editorial Committee of Annals of Thoracic and Cardiovascular Surgery. All rights reserved. 564
Ann Thorac Cardiovasc Surg Vol. 21, No. 6 (2015)
Bronchial Artery Aneurysm
(a)
** RML bronchus **
*
RLL bronchus
(b) Fig. 1 B ronchoscopy revealed a submucous elevated lesion (**) in the right middle bronchus. RML: right middle lobe; RLL: right lower lobe
A4 and A5 pulmonary artery (Fig. 3). First, we ligated and cut winding dilated bronchial artery at the level of carina in the posterior mediastinum. However, the pulsation of the BAA did not diminish and a thrill was remained. Next, we dissected BAA from pulmonary artery carefully. A small branch of A4 PA was separated, and the small vessel connecting to the BAA could be ligated. A5 PA was separated similarly, however BAA was ruptured not to identify the other small vessel connecting to the BAA in taping of A5 PA with forceps. After a clamp of the BAA, middle lobe lobectomy was performed. We removed the aneurysm with dilated bronchilal artery connecting to the aneurysm. We used autologous blood transfusion system during the operation. Intraoperative blood loss was 1000 ml without non-autologous transfusion. The postoperative course was uneventful, and the patient was discharged on the seventh postoperative day. The 35 days postoperative chest CT scans showed no evidence of BAA.
Discussion Bronchial artery aneurysm (BAA) is rare, detected in fewer than 1% of all patients who undergo selective bronchial arteriography.6) Bronchial artery aneurysm (BAA) can be classified according to the location as either mediastinal or intrapulmonary.7) The chief complaints for mediastinal BAA is relative to compression or rupture into contiguous structures. Intrapulmonary BAA presents with hemoptysis, which is the most frequent symptom. BAA in this case could not be classified either type because of hilar lesion with compression of right middle bronchus. Ann Thorac Cardiovasc Surg Vol. 21, No. 6 (2015)
A4 A5
*
Fig. 2 3 -D CT demonstrated an aneurysm (*) with racemose hemangioma (**) (a), and the aneurysm located between A4 and A5 connecting a narrow vessel (b).
It was located in the horizontal fissure of the right lung, and directly connected to the pulmonary artery, and presented no symptoms. BAA connecting directly to the proximal pulmonary artery is extremely rare. The bronchial artery arises mainly from thoracic aorta or its branches, and empties mainly into the pulmonary vein and a lesser bronchial vein system that enters the azygos venous system on the right.8) Existence of small anastomosis between the bronchial and pulmonary artery in the peripheral area were known.9) We thought the increasing blood flow of the bronchial artery caused to generate the aberrant anastomosis in this case. BAA in this case is not only anastomosed with pulmonary artery but also accompanied with racemose hemangioma. Racemose hemangioma of the bronchial arteries is a rare abnormality and is characterized by enlarged and convoluted bronchial arteries arranged segmentally along the longitudinal axis of bronchus. Since the term “racemose hemangioma” is firstly reported by v Babo H, et al. in Germany in 1976,10) some 565
Kitami A, et al.
have detected and ligated the aberrant dilated artery in the anterior side of the right main bronchus as possible as we can. Second, we could not detect the second aberrant small vessel connecting between BAA and pulmonary artery. If we had dissected the BAA more carefully after the ligation of first aberrant artery, we might remove the BAA without rupture.
RUL A4
* PA
Conclusion
BAA RML
**
RLL
A5
RUL
Disclosure Statements
A4
* RML BAA
There are no companies, etc. in a relation of conflict of interest requiring disclosure in relation to this manuscript.
References
PA A5
**
RLL Fig. 3 A small branch of A4 was separated, and the small vessel connecting to the BAA (*) could be ligated. A5 was separated similarly, however BAA was ruptured not to identify the other small vessel connecting to the BAA (**). BAA: bronchial artery aneurysm; RUL: right upper lobe; PA: pulmonary artery
literatures about racemose hemangioma reported in Japan. However this term seems to be used almost only in Japan, other case reports with same clinical findings were represented as “hypervascularity, dilatation and tortuosity of bronchial arteries”, “bronchial artery varices”, “arteriovenous malformation” so on. I think it is favorable to make a consensus of its definition among countries. BAA is potentially life threatening, so once the diagnosis is confirmed, the aneurysm should either be embolized using a transcatheter approach or resected surgically. Recently, transcatheter embolization has become more common. We performed aneurysmectomy with resection of racemose hemangioma in this case, because the aneurysm continued to have persistent flow in spite of two attempts at embolization. We reflect two points to prevent a rupture of the aneurysm during the operation. First, we could not reduce a blood flow of the aneurysm after the ligation of winding dilated bronchial artery at the level of carina in the posterior mediastinum. We ought to 566
We presented a surgical case of bronchial artery aneurysm connecting directly right pulmonary artery. In dissection of aneurysm, it is important to reduce a blood flow of the aneurysm with the ligation of winding dilated bronchial artery.
1) Kuwahara O, Takeda S, Dohi H, et al. A rare case of primary racemose hemangioma of the bronchial artery anastomosed with pulmonary artery. J Jpn Soc Bronchol 1998; 10: 170-4. 2) Shimokawaji T, Kobayashi H, Kawano S, et al. A case of idiopathic bronchial artery aneurysm connecting to the pulmonary artery. Nippon Kokyuki Gakkai Zasshi 2008; 46: 202-5. 3) Saito Y, Ueda Y, Imamura H, et al. Operative aneurysmectomy and middle lobectomy for asymptomatic bronchial artery aneurysm in young patient. Euro J Cardiothorac Surg 2000; 18: 366-9. 4) Cao DB, Yang SR, Pan RD, et al. Bronchial artery aneurysms and bronchial artery-pulmonary artery fistula. Euro J Cardiothorac Surg 2012; 42: e21. 5) Lin J, Wood DE. Bronchial artery aneurysm refractory to transcatheter embolization. Ann Thorac Surg 2008; 86: 306-8. 6) Fujita J, Akashi K, Kunikane H, et al. A case of bronchial artery aneurysm demonstrating a mass shadow on chest X-ray film. Nihon Kyobu Shikkan Gakkai Zasshi 1991; 29: 1591-5. 7) Osada H, Kawada T, Ashida H, et al. Bronchial artery aneurysm. Ann Thorac Surg 1986; 41: 440-2. 8) Shields TW. Surgical anatomy of the lungs. In: Shields TW, Locicero III J, Ponn RB, Rusch VW, eds.; General Thoracic Surgery 6th edition. Philadelphia, PA: Lippincott Williams & Wilkins 2005; pp 69-70. 9) PUMP KK. The bronchial arteries and their anastomoses in the human lung. Dis Chest 1963; 43: 245-55. 10) v Babo H, Huzly A, Deininger HK, et al. [Angiomas and angioma-like changes of the bronchial arteries (author’s transl)]. Rofo 1976; 124: 103-10. (in German) Ann Thorac Cardiovasc Surg Vol. 21, No. 6 (2015)
Case Report
Online June 5, 2015 doi: 10.5761/atcs.cr.15-00054
A Surgical Case of Bronchial Artery Aneurysm Directory Connecting with Pulmonary Artery Akihiko Kitami, MD,1 Fumitoshi Sano, MD,1 Shoko Hayashi, MD,1 Kosuke Suzuki, MD,1 Shugo Uematsu, MD,1 Takashi Suzuki, MD,1 and Noriyuki Saeki, MD2
We present a surgical case of bronchial artery aneurysm (BAA) connecting pulmonary artery accompanied with racemose hemangioma. This is a third surgical case report of BAA directly connecting pulmonary artery in the English literature. A 63-year-old female was found a BAA, 2 cm in diameter, connecting right A4 pulmonary artery. The patient underwent two attempts for embolization. However, due to extensive collaterals, there was persistent flow in the aneurysm. Standard lateral thoracotomy was performed. A BAA was located between A4 and A5 PA. A small branch of A4 PA was separated, and the small vessel connecting to the BAA could be ligated. A5 PA was separated similarly, however BAA was ruptured not to identify the other small vessel connecting to the BAA. After a clamp of the BAA, middle lobe lobectomy was performed. We removed the aneurysm with dilated bronchial artery connecting to the aneurysm. The postoperative course was uneventful. Keywords: bronchial artery aneurysm, connection to the pulmonary artery, racemose hemangioma
Introduction
Case Report
Bronchial artery aneurysm (BAA) is rare, but they are potentially life threatening and require treatment to avoid rupture and other complications. We present a surgical case of bronchial artery aneurysm connecting directly right pulmonary artery. To our knowledge, a few cases of bronchial artery aneurysm connecting pulmonary artery has been reported in Japanese literature,1,2) and only three in English literature.3–5) This is a third surgical case report of bronchial artery aneurysm directly connecting pulmonary artery in the English literature.
A 63-year-old female was found to have an abnormal shadow in a regular checkup chest X-ray film in 2005. She had no symptoms such as bloody sputum, hemoptysis or cardiac failure. Chest computed tomography (CT) scan revealed a round mass adjacent to the pulmonary artery in the right hilum, and bronchial artery dilated like vine; so called racemose hemangioma. Aortography, bronchial arteriography and pulmonary arteriography showed a bronchial artery aneurysm, 2 cm in diameter, connecting right pulmonary artery. The patient underwent two attempts for embolization at another hospital in 2006 and 2007. However, due to extensive collaterals, there was persistent flow in the aneurysm. The patient was referred to our hospital in 2011 for the purpose of surgical resection. Bronchoscopy revealed a submucous elevated lesion in the right middle bronchus (Fig. 1). Three dimensional computed tomography (3-D CT) demonstrated a narrow vessel which is connected aneurysm and right A4 pulmonary artery (Fig. 2). Standard lateral thoracotomy was performed via the fifth. A BAA 20 mm in diameter with thrill was located between
Respiratory Disease Center, Showa University Northern Yokohama Hospital, Yokohama, Kanagawa, Japan 2Department of Thoracic Surgery, Ofuna Chuo Hospital, Kamakura, Kanagawa, Japan 1
Received: February 17, 2015; Accepted: March 20, 2015 Corresponding author: Akihiko Kitami, MD. Respiratory Disease Center, Showa University Northern Yokohama Hospital, 35-1 Chigasaki– Chuo, Tsuzuki-ku, Yokohama, Kanagawa 224-0032, Japan E-mail: [email protected] ©2015 The Editorial Committee of Annals of Thoracic and Cardiovascular Surgery. All rights reserved. 564
Ann Thorac Cardiovasc Surg Vol. 21, No. 6 (2015)
Bronchial Artery Aneurysm
(a)
** RML bronchus **
*
RLL bronchus
(b) Fig. 1 B ronchoscopy revealed a submucous elevated lesion (**) in the right middle bronchus. RML: right middle lobe; RLL: right lower lobe
A4 and A5 pulmonary artery (Fig. 3). First, we ligated and cut winding dilated bronchial artery at the level of carina in the posterior mediastinum. However, the pulsation of the BAA did not diminish and a thrill was remained. Next, we dissected BAA from pulmonary artery carefully. A small branch of A4 PA was separated, and the small vessel connecting to the BAA could be ligated. A5 PA was separated similarly, however BAA was ruptured not to identify the other small vessel connecting to the BAA in taping of A5 PA with forceps. After a clamp of the BAA, middle lobe lobectomy was performed. We removed the aneurysm with dilated bronchilal artery connecting to the aneurysm. We used autologous blood transfusion system during the operation. Intraoperative blood loss was 1000 ml without non-autologous transfusion. The postoperative course was uneventful, and the patient was discharged on the seventh postoperative day. The 35 days postoperative chest CT scans showed no evidence of BAA.
Discussion Bronchial artery aneurysm (BAA) is rare, detected in fewer than 1% of all patients who undergo selective bronchial arteriography.6) Bronchial artery aneurysm (BAA) can be classified according to the location as either mediastinal or intrapulmonary.7) The chief complaints for mediastinal BAA is relative to compression or rupture into contiguous structures. Intrapulmonary BAA presents with hemoptysis, which is the most frequent symptom. BAA in this case could not be classified either type because of hilar lesion with compression of right middle bronchus. Ann Thorac Cardiovasc Surg Vol. 21, No. 6 (2015)
A4 A5
*
Fig. 2 3 -D CT demonstrated an aneurysm (*) with racemose hemangioma (**) (a), and the aneurysm located between A4 and A5 connecting a narrow vessel (b).
It was located in the horizontal fissure of the right lung, and directly connected to the pulmonary artery, and presented no symptoms. BAA connecting directly to the proximal pulmonary artery is extremely rare. The bronchial artery arises mainly from thoracic aorta or its branches, and empties mainly into the pulmonary vein and a lesser bronchial vein system that enters the azygos venous system on the right.8) Existence of small anastomosis between the bronchial and pulmonary artery in the peripheral area were known.9) We thought the increasing blood flow of the bronchial artery caused to generate the aberrant anastomosis in this case. BAA in this case is not only anastomosed with pulmonary artery but also accompanied with racemose hemangioma. Racemose hemangioma of the bronchial arteries is a rare abnormality and is characterized by enlarged and convoluted bronchial arteries arranged segmentally along the longitudinal axis of bronchus. Since the term “racemose hemangioma” is firstly reported by v Babo H, et al. in Germany in 1976,10) some 565
Kitami A, et al.
have detected and ligated the aberrant dilated artery in the anterior side of the right main bronchus as possible as we can. Second, we could not detect the second aberrant small vessel connecting between BAA and pulmonary artery. If we had dissected the BAA more carefully after the ligation of first aberrant artery, we might remove the BAA without rupture.
RUL A4
* PA
Conclusion
BAA RML
**
RLL
A5
RUL
Disclosure Statements
A4
* RML BAA
There are no companies, etc. in a relation of conflict of interest requiring disclosure in relation to this manuscript.
References
PA A5
**
RLL Fig. 3 A small branch of A4 was separated, and the small vessel connecting to the BAA (*) could be ligated. A5 was separated similarly, however BAA was ruptured not to identify the other small vessel connecting to the BAA (**). BAA: bronchial artery aneurysm; RUL: right upper lobe; PA: pulmonary artery
literatures about racemose hemangioma reported in Japan. However this term seems to be used almost only in Japan, other case reports with same clinical findings were represented as “hypervascularity, dilatation and tortuosity of bronchial arteries”, “bronchial artery varices”, “arteriovenous malformation” so on. I think it is favorable to make a consensus of its definition among countries. BAA is potentially life threatening, so once the diagnosis is confirmed, the aneurysm should either be embolized using a transcatheter approach or resected surgically. Recently, transcatheter embolization has become more common. We performed aneurysmectomy with resection of racemose hemangioma in this case, because the aneurysm continued to have persistent flow in spite of two attempts at embolization. We reflect two points to prevent a rupture of the aneurysm during the operation. First, we could not reduce a blood flow of the aneurysm after the ligation of winding dilated bronchial artery at the level of carina in the posterior mediastinum. We ought to 566
We presented a surgical case of bronchial artery aneurysm connecting directly right pulmonary artery. In dissection of aneurysm, it is important to reduce a blood flow of the aneurysm with the ligation of winding dilated bronchial artery.
1) Kuwahara O, Takeda S, Dohi H, et al. A rare case of primary racemose hemangioma of the bronchial artery anastomosed with pulmonary artery. J Jpn Soc Bronchol 1998; 10: 170-4. 2) Shimokawaji T, Kobayashi H, Kawano S, et al. A case of idiopathic bronchial artery aneurysm connecting to the pulmonary artery. Nippon Kokyuki Gakkai Zasshi 2008; 46: 202-5. 3) Saito Y, Ueda Y, Imamura H, et al. Operative aneurysmectomy and middle lobectomy for asymptomatic bronchial artery aneurysm in young patient. Euro J Cardiothorac Surg 2000; 18: 366-9. 4) Cao DB, Yang SR, Pan RD, et al. Bronchial artery aneurysms and bronchial artery-pulmonary artery fistula. Euro J Cardiothorac Surg 2012; 42: e21. 5) Lin J, Wood DE. Bronchial artery aneurysm refractory to transcatheter embolization. Ann Thorac Surg 2008; 86: 306-8. 6) Fujita J, Akashi K, Kunikane H, et al. A case of bronchial artery aneurysm demonstrating a mass shadow on chest X-ray film. Nihon Kyobu Shikkan Gakkai Zasshi 1991; 29: 1591-5. 7) Osada H, Kawada T, Ashida H, et al. Bronchial artery aneurysm. Ann Thorac Surg 1986; 41: 440-2. 8) Shields TW. Surgical anatomy of the lungs. In: Shields TW, Locicero III J, Ponn RB, Rusch VW, eds.; General Thoracic Surgery 6th edition. Philadelphia, PA: Lippincott Williams & Wilkins 2005; pp 69-70. 9) PUMP KK. The bronchial arteries and their anastomoses in the human lung. Dis Chest 1963; 43: 245-55. 10) v Babo H, Huzly A, Deininger HK, et al. [Angiomas and angioma-like changes of the bronchial arteries (author’s transl)]. Rofo 1976; 124: 103-10. (in German) Ann Thorac Cardiovasc Surg Vol. 21, No. 6 (2015)
