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Hemomediastinum Caused by Rupture of a Bronchial Artery Aneurysm: Successful Treatment by Embolization with N-Butyl-2-Cyanoacrylate

2001; Elsevier BV; Volume: 12; Issue: 11 Linguagem: Inglês

10.1016/s1051-0443(07)61563-2

1535-7732

Mauro Pugnale, François Portier, André Lamarre, Nermin Halkic, Hans-Beat Riis, Stéphan Wicky, P. Schnyder, Alban Denys,

Case Reports on Hematomas

Editor:Acute hemomediastinum and hemothorax are usually related to chest trauma, rupture of thoracic aortic aneurysm, or aortic dissection. Causes of spontaneous mediastinal hemorrhage have been subdivided by Ellison et al (1Ellison RT Carrao WM Fox MJ Braman SS Spontaneous mediastinal hemorrhage in patients on chronic hemodialysis.Ann Intern Med. 1981; 95: 704-706Crossref PubMed Scopus (19) Google Scholar) in four categories: (i) complication of enlarging mediastinal masses, (ii) transient increase in intrathoracic pressure, (iii) sudden sustained hypertension, and (iv) altered hemostasis (1Ellison RT Carrao WM Fox MJ Braman SS Spontaneous mediastinal hemorrhage in patients on chronic hemodialysis.Ann Intern Med. 1981; 95: 704-706Crossref PubMed Scopus (19) Google Scholar, 2Gattoni F Tagliaferri B Boioli F Tonolini M Uslenghi CM One case of ruptured bronchial artery aneurysm studied with spiral CT and literature review.Radiol Med (Torino). 1999; 98: 418-421PubMed Google Scholar). Rupture of a bronchial artery aneurysm (BAA) is rarely the etiology of mediastinal hemorrhage. We present a case of a ruptured BAA in which the diagnosis was made by thoracic computed tomography (CT) and successful treatment was achieved by embolization with n-butyl-2-cyanoacrylate. It allows occlusion of feeding and outflow vessels as reported with other embolization materials (3Sakai T Razavi MK Semba CP Kee ST Sze DY Dake MD Percutaneous treatment of bronchial artery aneurysm with use of transcatheter coil embolization and thoracic aortic stent-graft placement.J Vasc Interv Radiol. 1998; 9: 1025-1028Abstract Full Text PDF PubMed Scopus (28) Google Scholar).Clinical manifestations, causes, and treatments of BAA will be discussed.A 72-year-old man with a clinical history of chronic obstructive pulmonary disease presented at the emergency room reporting sudden radiating epigastric pain. At arrival, his hemoglobin level was 11.5 g/L and decreased 2 g/L during the 6 first hours after admission. A thoracic plain radiograph revealed an enlarged mediastinum. Plain CT (Lightspeed; General Electric, Milwaukee, WI) showed an enlarged mediastinum. After injection, enlarged bronchial arteries (3 mm in diameter) were visible with a hemomediastinum and a nodular enhancing homogenous mass of 1.5 cm diameter just below the carina level, which was suspected to be a BAA (Fig 1a). Bronchial arteriography arteries was then performed. A large anterior bronchial common trunk was catheterized with use of a 5-F Cobra 2 catheter (Terumo, Tokyo, Japan). With use of a coaxial 3-F catheter (Terumo), the origin of the right bronchial artery was catheterized and, on angiographic opacification, we confirmed a single right bronchial artery aneurysm (1.5 cm in diameter). Catheterization of the efferent branches could not be performed because of vessel tortuosity (Fig 1b); three different microcatheters and a 0.014-inch guide wire were used without success. As a consequence, we decided to occlude the aneurysm and the efferent branches with use of embucrylate (Histoacryl, B. Braun, Melsungen, Germany) mixed with iodized oil (Lipiodol; Guerbet, Aulnay-sous-Bois, France). Injection of 0.3 mL (50% Histoacryl/50% Lipiodol) was necessary to allow a complete occlusion of the aneurysm (Fig 1c). One week later, the patient was discharged and CT performed 1 month after discharge confirmed almost total regression of the hemomediastinum and no enhancement of the aneurysm in early and delayed vascular enhancement phases (Fig 1d).Bronchial artery aneurysm is a rare entity. It can be found incidentally or be related to dramatic hemorrhagic manifestations if a rupture happens. To our knowledge, 33 cases of BAA have been reported in the literature (1Ellison RT Carrao WM Fox MJ Braman SS Spontaneous mediastinal hemorrhage in patients on chronic hemodialysis.Ann Intern Med. 1981; 95: 704-706Crossref PubMed Scopus (19) Google Scholar, 2Gattoni F Tagliaferri B Boioli F Tonolini M Uslenghi CM One case of ruptured bronchial artery aneurysm studied with spiral CT and literature review.Radiol Med (Torino). 1999; 98: 418-421PubMed Google Scholar, 3Sakai T Razavi MK Semba CP Kee ST Sze DY Dake MD Percutaneous treatment of bronchial artery aneurysm with use of transcatheter coil embolization and thoracic aortic stent-graft placement.J Vasc Interv Radiol. 1998; 9: 1025-1028Abstract Full Text PDF PubMed Scopus (28) Google Scholar, 4Sancho 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: 1122-1126Abstract Full Text PDF PubMed Scopus (21) Google Scholar, 5Braks E Pauleit D Strunk H Schild H Diagnosis and therapy of bronchial artery aneurysm.Rofo Fortschr Geb Rontgenstr Neuen Bildgeb Verfahr. 1999; 170: 123-124Crossref Scopus (4) Google Scholar, 6Servois V Denys A Silbert A Mycotic aneurysm of the bronchial artery: a rare cause of hemoptysis.AJR Am J Roentgenol. 1992; 159: 428Crossref PubMed Scopus (11) Google Scholar, 7Ishizaki N Shimokawa S Tanaka K et al.Ruptured bronchial artery aneurysm associated with pleural telangiectasis and tortuous portal obstruction: report of a case.Surg Today. 1995; 25: 852-854Crossref PubMed Scopus (21) Google Scholar, 8Remy-Jardin M Remy J Ramon P Fellous G Mediastinal bronchial artery aneurysm: dynamic computed tomography appearance.Cardiovasc Intervent Radiol. 1991; 14: 118-210Crossref PubMed Scopus (33) Google Scholar). Of those 33, one third were asymptomatic at diagnosis and the most frequent clinical sign was hemoptysis (39%). Other symptoms are variable and are related to compression effect or rupture into adjacent organs (esophagus, superior vena cava, main bronchus). Ruptured BAA rarely presents as a spontaneous hemomediastinum as in our case (5Braks E Pauleit D Strunk H Schild H Diagnosis and therapy of bronchial artery aneurysm.Rofo Fortschr Geb Rontgenstr Neuen Bildgeb Verfahr. 1999; 170: 123-124Crossref Scopus (4) Google Scholar).BAAs are usually a consequence of bronchiectasis or recurrent bronchopulmonary inflammation. It is hypothesized that increased bronchial arterial flow to the lungs during inflammation could be a mechanism of bronchial artery dilation and subsequent aneurysm formation. Mediastinal BAA can be incidentally identified or suspected on CT, but in a hemodynamically stable patient, the definite diagnosis has to be confirmed by selective bronchial artery angiography, which enables therapeutic embolization at the same time. Our case is the second to be reported of a ruptured BAA treated by embolization. The other one was described by Ishisaki et al (7Ishizaki N Shimokawa S Tanaka K et al.Ruptured bronchial artery aneurysm associated with pleural telangiectasis and tortuous portal obstruction: report of a case.Surg Today. 1995; 25: 852-854Crossref PubMed Scopus (21) Google Scholar) and was embolized preoperatively. Most cases described did not involve CT and were treated immediately in the operating room after the discovery of an enlarged mediastinum on a chest plain radiograph. Our case stresses the utility of CT with contrast injection. This exploration eliminates other potential causes of hemomediastinum such as aortic aneurysm or hemorrhagic mediastinal mass and even allows a suspected diagnosis of bronchial artery aneurysm.Remy-Jardin et al (8Remy-Jardin M Remy J Ramon P Fellous G Mediastinal bronchial artery aneurysm: dynamic computed tomography appearance.Cardiovasc Intervent Radiol. 1991; 14: 118-210Crossref PubMed Scopus (33) Google Scholar) showed that transcatheter embolization was successful in obliterating the blood flow and excluding the aneurysm if the segment between the aneurysm and aorta is judged sufficiently long on bronchial arteriography. If this segment is short, as recommended by Sakai (3Sakai T Razavi MK Semba CP Kee ST Sze DY Dake MD Percutaneous treatment of bronchial artery aneurysm with use of transcatheter coil embolization and thoracic aortic stent-graft placement.J Vasc Interv Radiol. 1998; 9: 1025-1028Abstract Full Text PDF PubMed Scopus (28) Google Scholar), the origin of the bronchial arteries can be covered with a covered stent after the outflow vessels are occluded. In the literature, different embolic materials have been used, such as steel coils, gelatin particles, and occlusion balloons. Whatever embolic material is used, most authors put the emphasis on the necessity to occlude not only the feeding vessel but also efferent branches to avoid retrograde filling of the aneurysm. In our case, catheterization of the efferent branches was impossible because of the tortuosity of the right bronchial artery. When embucrylate is adequately diluted, it casts distally to the injection site and allows occlusion of distal vessels. In our case, no intercostal branches were present, so there was no risk of inadvertent embolization of spinal branches. If intercostal branches werepresent, we probably would have occluded the feeding artery with coils. Surgical treatment has been also proposed for ruptured BAA. Because of higher cost, longer hospital stay, and potential associated complications, it seems logical to attempt embolization first and to withhold surgery for situations when an associated procedure is needed—for example, a lobectomy for dilated bronchi or when embolization cannot be performed.If a patient is hemodynamically stable, embolization seems an efficient alternative to surgery to treat BAA. N-butyl-2-cyanoacrylate can be used for such embolization. This embolic material occludes feeding vessels and collateral efferent vessels, avoiding retrograde filling of the aneurysm. Because of lack of control of cast formation, there is a risk of distal embolization when intercostal branches come from the right bronchial artery. Editor: Acute hemomediastinum and hemothorax are usually related to chest trauma, rupture of thoracic aortic aneurysm, or aortic dissection. Causes of spontaneous mediastinal hemorrhage have been subdivided by Ellison et al (1Ellison RT Carrao WM Fox MJ Braman SS Spontaneous mediastinal hemorrhage in patients on chronic hemodialysis.Ann Intern Med. 1981; 95: 704-706Crossref PubMed Scopus (19) Google Scholar) in four categories: (i) complication of enlarging mediastinal masses, (ii) transient increase in intrathoracic pressure, (iii) sudden sustained hypertension, and (iv) altered hemostasis (1Ellison RT Carrao WM Fox MJ Braman SS Spontaneous mediastinal hemorrhage in patients on chronic hemodialysis.Ann Intern Med. 1981; 95: 704-706Crossref PubMed Scopus (19) Google Scholar, 2Gattoni F Tagliaferri B Boioli F Tonolini M Uslenghi CM One case of ruptured bronchial artery aneurysm studied with spiral CT and literature review.Radiol Med (Torino). 1999; 98: 418-421PubMed Google Scholar). Rupture of a bronchial artery aneurysm (BAA) is rarely the etiology of mediastinal hemorrhage. We present a case of a ruptured BAA in which the diagnosis was made by thoracic computed tomography (CT) and successful treatment was achieved by embolization with n-butyl-2-cyanoacrylate. It allows occlusion of feeding and outflow vessels as reported with other embolization materials (3Sakai T Razavi MK Semba CP Kee ST Sze DY Dake MD Percutaneous treatment of bronchial artery aneurysm with use of transcatheter coil embolization and thoracic aortic stent-graft placement.J Vasc Interv Radiol. 1998; 9: 1025-1028Abstract Full Text PDF PubMed Scopus (28) Google Scholar).Clinical manifestations, causes, and treatments of BAA will be discussed. A 72-year-old man with a clinical history of chronic obstructive pulmonary disease presented at the emergency room reporting sudden radiating epigastric pain. At arrival, his hemoglobin level was 11.5 g/L and decreased 2 g/L during the 6 first hours after admission. A thoracic plain radiograph revealed an enlarged mediastinum. Plain CT (Lightspeed; General Electric, Milwaukee, WI) showed an enlarged mediastinum. After injection, enlarged bronchial arteries (3 mm in diameter) were visible with a hemomediastinum and a nodular enhancing homogenous mass of 1.5 cm diameter just below the carina level, which was suspected to be a BAA (Fig 1a). Bronchial arteriography arteries was then performed. A large anterior bronchial common trunk was catheterized with use of a 5-F Cobra 2 catheter (Terumo, Tokyo, Japan). With use of a coaxial 3-F catheter (Terumo), the origin of the right bronchial artery was catheterized and, on angiographic opacification, we confirmed a single right bronchial artery aneurysm (1.5 cm in diameter). Catheterization of the efferent branches could not be performed because of vessel tortuosity (Fig 1b); three different microcatheters and a 0.014-inch guide wire were used without success. As a consequence, we decided to occlude the aneurysm and the efferent branches with use of embucrylate (Histoacryl, B. Braun, Melsungen, Germany) mixed with iodized oil (Lipiodol; Guerbet, Aulnay-sous-Bois, France). Injection of 0.3 mL (50% Histoacryl/50% Lipiodol) was necessary to allow a complete occlusion of the aneurysm (Fig 1c). One week later, the patient was discharged and CT performed 1 month after discharge confirmed almost total regression of the hemomediastinum and no enhancement of the aneurysm in early and delayed vascular enhancement phases (Fig 1d). Bronchial artery aneurysm is a rare entity. It can be found incidentally or be related to dramatic hemorrhagic manifestations if a rupture happens. To our knowledge, 33 cases of BAA have been reported in the literature (1Ellison RT Carrao WM Fox MJ Braman SS Spontaneous mediastinal hemorrhage in patients on chronic hemodialysis.Ann Intern Med. 1981; 95: 704-706Crossref PubMed Scopus (19) Google Scholar, 2Gattoni F Tagliaferri B Boioli F Tonolini M Uslenghi CM One case of ruptured bronchial artery aneurysm studied with spiral CT and literature review.Radiol Med (Torino). 1999; 98: 418-421PubMed Google Scholar, 3Sakai T Razavi MK Semba CP Kee ST Sze DY Dake MD Percutaneous treatment of bronchial artery aneurysm with use of transcatheter coil embolization and thoracic aortic stent-graft placement.J Vasc Interv Radiol. 1998; 9: 1025-1028Abstract Full Text PDF PubMed Scopus (28) Google Scholar, 4Sancho 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: 1122-1126Abstract Full Text PDF PubMed Scopus (21) Google Scholar, 5Braks E Pauleit D Strunk H Schild H Diagnosis and therapy of bronchial artery aneurysm.Rofo Fortschr Geb Rontgenstr Neuen Bildgeb Verfahr. 1999; 170: 123-124Crossref Scopus (4) Google Scholar, 6Servois V Denys A Silbert A Mycotic aneurysm of the bronchial artery: a rare cause of hemoptysis.AJR Am J Roentgenol. 1992; 159: 428Crossref PubMed Scopus (11) Google Scholar, 7Ishizaki N Shimokawa S Tanaka K et al.Ruptured bronchial artery aneurysm associated with pleural telangiectasis and tortuous portal obstruction: report of a case.Surg Today. 1995; 25: 852-854Crossref PubMed Scopus (21) Google Scholar, 8Remy-Jardin M Remy J Ramon P Fellous G Mediastinal bronchial artery aneurysm: dynamic computed tomography appearance.Cardiovasc Intervent Radiol. 1991; 14: 118-210Crossref PubMed Scopus (33) Google Scholar). Of those 33, one third were asymptomatic at diagnosis and the most frequent clinical sign was hemoptysis (39%). Other symptoms are variable and are related to compression effect or rupture into adjacent organs (esophagus, superior vena cava, main bronchus). Ruptured BAA rarely presents as a spontaneous hemomediastinum as in our case (5Braks E Pauleit D Strunk H Schild H Diagnosis and therapy of bronchial artery aneurysm.Rofo Fortschr Geb Rontgenstr Neuen Bildgeb Verfahr. 1999; 170: 123-124Crossref Scopus (4) Google Scholar). BAAs are usually a consequence of bronchiectasis or recurrent bronchopulmonary inflammation. It is hypothesized that increased bronchial arterial flow to the lungs during inflammation could be a mechanism of bronchial artery dilation and subsequent aneurysm formation. Mediastinal BAA can be incidentally identified or suspected on CT, but in a hemodynamically stable patient, the definite diagnosis has to be confirmed by selective bronchial artery angiography, which enables therapeutic embolization at the same time. Our case is the second to be reported of a ruptured BAA treated by embolization. The other one was described by Ishisaki et al (7Ishizaki N Shimokawa S Tanaka K et al.Ruptured bronchial artery aneurysm associated with pleural telangiectasis and tortuous portal obstruction: report of a case.Surg Today. 1995; 25: 852-854Crossref PubMed Scopus (21) Google Scholar) and was embolized preoperatively. Most cases described did not involve CT and were treated immediately in the operating room after the discovery of an enlarged mediastinum on a chest plain radiograph. Our case stresses the utility of CT with contrast injection. This exploration eliminates other potential causes of hemomediastinum such as aortic aneurysm or hemorrhagic mediastinal mass and even allows a suspected diagnosis of bronchial artery aneurysm. Remy-Jardin et al (8Remy-Jardin M Remy J Ramon P Fellous G Mediastinal bronchial artery aneurysm: dynamic computed tomography appearance.Cardiovasc Intervent Radiol. 1991; 14: 118-210Crossref PubMed Scopus (33) Google Scholar) showed that transcatheter embolization was successful in obliterating the blood flow and excluding the aneurysm if the segment between the aneurysm and aorta is judged sufficiently long on bronchial arteriography. If this segment is short, as recommended by Sakai (3Sakai T Razavi MK Semba CP Kee ST Sze DY Dake MD Percutaneous treatment of bronchial artery aneurysm with use of transcatheter coil embolization and thoracic aortic stent-graft placement.J Vasc Interv Radiol. 1998; 9: 1025-1028Abstract Full Text PDF PubMed Scopus (28) Google Scholar), the origin of the bronchial arteries can be covered with a covered stent after the outflow vessels are occluded. In the literature, different embolic materials have been used, such as steel coils, gelatin particles, and occlusion balloons. Whatever embolic material is used, most authors put the emphasis on the necessity to occlude not only the feeding vessel but also efferent branches to avoid retrograde filling of the aneurysm. In our case, catheterization of the efferent branches was impossible because of the tortuosity of the right bronchial artery. When embucrylate is adequately diluted, it casts distally to the injection site and allows occlusion of distal vessels. In our case, no intercostal branches were present, so there was no risk of inadvertent embolization of spinal branches. If intercostal branches werepresent, we probably would have occluded the feeding artery with coils. Surgical treatment has been also proposed for ruptured BAA. Because of higher cost, longer hospital stay, and potential associated complications, it seems logical to attempt embolization first and to withhold surgery for situations when an associated procedure is needed—for example, a lobectomy for dilated bronchi or when embolization cannot be performed. If a patient is hemodynamically stable, embolization seems an efficient alternative to surgery to treat BAA. N-butyl-2-cyanoacrylate can be used for such embolization. This embolic material occludes feeding vessels and collateral efferent vessels, avoiding retrograde filling of the aneurysm. Because of lack of control of cast formation, there is a risk of distal embolization when intercostal branches come from the right bronchial artery.