Cardiovasc Intervent Radiol DOI 10.1007/s00270-014-1007-x
LETTER TO THE EDITOR
Embolization of a Giant Intercostal Aneurysm and Arteriovenous Malformation Lawrence Bonne • Piet Vanhoenacker Luc Defreyne
•
Received: 29 May 2014 / Accepted: 1 September 2014 Ó Springer Science+Business Media New York and the Cardiovascular and Interventional Radiological Society of Europe (CIRSE) 2014
Introduction Intercostal aneurysms, arteriovenous malformations (AVM) and fistulas (AVF) are rare lesions. Only a few cases have been described in the literature [1–10]. Most lesions are considered to be acquired, i.e., secondary to trauma or iatrogenic, although some cases may be congenital in origin [1, 6–8, 10]. We present a case of a giant intercostal artery aneurysm associated with a paravertebral pleural arteriovenous malformation in a young patient, which was treated by embolization with coils and glue, using a bilateral transarterial approach.
Case Report A 41-year-old male patient presented to our hospital for routine follow-up of pneumonia. In the previous years, the patient had suffered from two spontaneous bilateral pneumothoraces which were treated by drainage. Physical examination was unremarkable. Chest auscultation was normal. Chest radiography showed a mass in the posterior mediastinum. Further investigation with MRI and endoscopic ultrasound revealed a large vascular lesion on the L. Bonne (&) P. Vanhoenacker Department of Radiology, OLV Hospital Aalst-Asse-Ninove, Moorselbaan 164, Aalst 9300, Belgium e-mail: [email protected] P. Vanhoenacker e-mail: [email protected] P. Vanhoenacker L. Defreyne Department of Vascular and Interventional Radiology, University Hospital Ghent, Ghent, Belgium e-mail: [email protected]
right side of the proximal descending thoracic aorta, which was thought to be a giant aneurysm of an intercostal artery, associated with a paravertebral, pleural arteriovenous malformation. Diagnostic angiography confirmed a postostial saccular aneurysm of the fourth, right intercostal artery, measuring approximately 8 cm (Fig. 1). The aneurysm communicated with a complex pleural AVM consisting of multiple nidal components and several draining pulmonary veins with an abnormal morphology (Fig. 1). Additional arterial feeders originated from the right subclavian artery and the third right intercostal artery. Further diagnostic work-up revealed no spinal arteries arising from the feeding arteries. The Adamkiewicz artery was confirmed to originate from an intercostobronchial artery on the left side. The lesion was thought to be related to thoracic drainage for a spontaneous pneumothorax the patient had suffered from years ago. Although the patient had no actual physical discomfort, treatment of the aneurysm was considered mandatory because of the high risk of growth and rupture of the aneurysm. Endovascular treatment was chosen, using a bilateral transarterial approach. The procedure was performed under general anesthesia. A 5 Fr and 6 Fr sheath were placed in the right and left femoral artery, respectively. A 5 Fr Cobra catheter was then placed in the proximal fourth right intercostal artery. Subsequently, the distal intercostal artery (outflow of the aneurysm) was catheterized using a 2.8 Fr Progreat microcatheter (Terumo, Leuven, Belgium) (Fig. 1C). A 4 Fr Cobra catheter was placed inside the aneurysm via the right femoral artery. The lateral part of the aneurysm was then embolized via the 4 Fr Cobra using 23 0.035 MReye stainless steel coils with diameter ranging from 2 to 5 mm (Cook, Denmark) (Fig. 2A). Next, the aneurysm outflow and therefore several arteriovenous fistulas were embolized via the microcatheter using a Glubran2
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L. Bonne et al.: Embolization of a Giant Intercostal Aneurysm and Arteriovenous Malformation
Fig. 1 A–C Selective angiogram of the fourth right intercostal artery in early arterial (A) and late arterial (B) phase revealing a giant postostial aneurysm connected with a pleural arteriovenous malformation.
Superselective angiogram of the aneurysm (C) shows abnormal arteries draining into pulmonary veins
Fig. 2 A–B Angiogram after coil embolization of the lateral and middle part of the aneurysm (A). The medial part is still patent and is now small enough to be embolized with glue via the microcatheter.
Control angiogram of the thoracic aorta (B) shows occlusion of the aneurysm and residual opacification of the cranial part of the AVM
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L. Bonne et al.: Embolization of a Giant Intercostal Aneurysm and Arteriovenous Malformation
Fig. 3 A–C Control angiogram of the thoracic aorta after 6 months shows occlusion of the aneurysm (with coil mass) and residual opacification of the AVM
(GEM, Italy)/Lipiodol (Guerbet, France) mixture (1:3) under fluoroscopic control. The 4 Fr Cobra catheter was then removed. Further Glubran2/Lipiodol embolization of the aneurysm was performed while slowly pulling back the microcatheter, until the aneurysm inflow and the proximal part of the intercostal artery were closed. Subsequent angiograms of the thoracic aorta and the nearby intercostal arteries showed occlusion of the aneurysm and part of the pleural arteriovenous malformation (Fig. 2B). There were no complications. The patient was discharged from the hospital 3 days later. A 6-month control angiography of the thoracic aorta confirmed the occlusion of the intercostal aneurysm. Part of the pleural AVM was still supplied by other intercostal and thoracic arteries (Fig. 3).
Discussion Intercostal artery aneurysms and AVM have been described, but are rare. Most reported intercostal AVM are
thought to be secondary to trauma, interventional procedures such as biopsy or transthoracic surgery or inflammatory pleural disease. Only a couple of cases were treated using an exclusively endovascular approach. In this case report, we describe a giant intercostal artery aneurysm associated with a pleural arteriovenous malformation in a young patient who had suffered from bilateral spontaneous pneumothoraces in the previous years. Thoracic drainage of these pneumothoraces is thought to be the cause of development of the lesion, although the etiology of the giant aneurysm is not entirely clear. A primary traumatic cause is possible. Its origin, however, might be flow-related as well, analogous to the flow-related aneurysms seen in intracranial AVM. To our knowledge, this is the first case of a giant aneurysm of the intercostal artery associated with a diffuse pleural arteriovenous malformation. For this reason, and because of the general rarity of these lesions, this case was considered a therapeutic challenge. The aim of the endovascular procedure was filling the aneurysm with coils, occluding the outflow and inflow of
123
L. Bonne et al.: Embolization of a Giant Intercostal Aneurysm and Arteriovenous Malformation
the aneurysm with diluted glue while also embolizing the part of the pleural AVM supplied by the intercostal artery. We combined coil and glue embolization to increase the maximum occlusive effect and to avoid retrograde vascularization via arteries branching from the aneurysmal sac. Therefore, two catheter systems were placed, one inside the aneurysm and one in the outflow. By injecting diluted glue in the outflow, we occluded parts of the pleural AVM. After the aneurysm was heavily reduced by the coil mass, the outflow, the residual aneurysmal sac and the inflow of the intercostal artery could be occluded by slow injection of glue through the microcatheter during its withdrawal. In this way, we sealed the aneurysm in the long term, as was confirmed on a control angiography after 6 months. We instated this waiting time of 6 months for control angiography to allow potentially missed or occult feeders to hypertrophy and become angiographically detectable, as well as to allow coil compaction to occur. The residual pleural AVM was not treated because it is at the time asymptomatic. Moreover, the extension of the pleural AVM and the multiplicity of the feeders would be a real endovascular challenge. It is important to emphasize that in case of a lesion involving the descending thoracic aorta and the intercostal arteries, spinal vascularization should be visualized before proceeding to any therapeutic procedure, in order to prevent unintended spinal cord ischemia. The intercostal arteries give rise to several vessels that supply the spinal cord, with the dominant thoracic anterior radiculary or ‘Adamkiewicz’ artery mostly originating on the left side at level T9–T12. In our case, diagnostic angiography did not reveal any spinal arteries arising from the pathological intercostal artery, and since the Adamkiewicz artery
123
originated from an intercostobronchial artery on the left side, endovascular treatment seemed safe and appropriate. Conflict of interest of interest.
The authors declare that they have no conflict
References 1. Rivera PP, Kole MK, Pelz DM et al (2006) Congenital intercostal arteriovenous malformation. AJR 187:W503–W506 2. Coulter TD, Maurer JR, Miller MT et al (1999) Chest wall arteriovenous fistula: an unusual complication after chest tube placement. Ann Thorac Surg 67:849–850 3. Derdeyn CP, Middleton WD, Allen BT, Nordlicht SM (1993) Acquired intercostal arteriovenous fistula: color Doppler ultrasonographic diagnosis. J Ultrasound Med 12:679–681 4. Lai JH, Yan HC, Kao SJ et al (1990) Intercostal arteriovenous fistula due to pleural biopsy. Thorax 45:976–978 5. Siddhartha W, Parmar H, Shrivastav M et al (2000) Endovascular glue embolization of an intercostal arteriovenous fistula: a nonsurgical option. J Postgrad Med 46:213–214 6. Yamasaki N, Hata H, Kusaga M, Kubo K (1977) A surgical case of congenital intercostal arteriovenous fistula. Nippon Kyobu Geka Gakkai Zasshi 25:936–939 7. Saito A, Takahashi T, Ezura M, Tominaga T (2007) Intercostal arteriovenous fistula associated with neurofibromatosis manifesting as congestive myelopathy: case report. Neurosurgery 61:E656–E657 8. Hamilton MCK, Occleshaw CJ, Calder AL (2005) Congenital arteriovenous fistula between an intercostal artery and the left brachiocephalic vein. Cardiol Young 15:437–438 9. Gonzalez R, Santolaya R, Prats R, Rodriguez P (2010) Left intercostal aneurysm fistulised to azygos vein as a late complication of thoracic trauma. Eur J Cardiothorac Surg 39:276 10. Kim HJ, Seon HJ, Choi S, Jang NK (2011) Ruptured aneurysm of intercostal artery aneurysm associated with Neurofibromatosis Type 1: a case report. Cardiovasc Interv Radiol 34:S74–S77
LETTER TO THE EDITOR
Embolization of a Giant Intercostal Aneurysm and Arteriovenous Malformation Lawrence Bonne • Piet Vanhoenacker Luc Defreyne
•
Received: 29 May 2014 / Accepted: 1 September 2014 Ó Springer Science+Business Media New York and the Cardiovascular and Interventional Radiological Society of Europe (CIRSE) 2014
Introduction Intercostal aneurysms, arteriovenous malformations (AVM) and fistulas (AVF) are rare lesions. Only a few cases have been described in the literature [1–10]. Most lesions are considered to be acquired, i.e., secondary to trauma or iatrogenic, although some cases may be congenital in origin [1, 6–8, 10]. We present a case of a giant intercostal artery aneurysm associated with a paravertebral pleural arteriovenous malformation in a young patient, which was treated by embolization with coils and glue, using a bilateral transarterial approach.
Case Report A 41-year-old male patient presented to our hospital for routine follow-up of pneumonia. In the previous years, the patient had suffered from two spontaneous bilateral pneumothoraces which were treated by drainage. Physical examination was unremarkable. Chest auscultation was normal. Chest radiography showed a mass in the posterior mediastinum. Further investigation with MRI and endoscopic ultrasound revealed a large vascular lesion on the L. Bonne (&) P. Vanhoenacker Department of Radiology, OLV Hospital Aalst-Asse-Ninove, Moorselbaan 164, Aalst 9300, Belgium e-mail: [email protected] P. Vanhoenacker e-mail: [email protected] P. Vanhoenacker L. Defreyne Department of Vascular and Interventional Radiology, University Hospital Ghent, Ghent, Belgium e-mail: [email protected]
right side of the proximal descending thoracic aorta, which was thought to be a giant aneurysm of an intercostal artery, associated with a paravertebral, pleural arteriovenous malformation. Diagnostic angiography confirmed a postostial saccular aneurysm of the fourth, right intercostal artery, measuring approximately 8 cm (Fig. 1). The aneurysm communicated with a complex pleural AVM consisting of multiple nidal components and several draining pulmonary veins with an abnormal morphology (Fig. 1). Additional arterial feeders originated from the right subclavian artery and the third right intercostal artery. Further diagnostic work-up revealed no spinal arteries arising from the feeding arteries. The Adamkiewicz artery was confirmed to originate from an intercostobronchial artery on the left side. The lesion was thought to be related to thoracic drainage for a spontaneous pneumothorax the patient had suffered from years ago. Although the patient had no actual physical discomfort, treatment of the aneurysm was considered mandatory because of the high risk of growth and rupture of the aneurysm. Endovascular treatment was chosen, using a bilateral transarterial approach. The procedure was performed under general anesthesia. A 5 Fr and 6 Fr sheath were placed in the right and left femoral artery, respectively. A 5 Fr Cobra catheter was then placed in the proximal fourth right intercostal artery. Subsequently, the distal intercostal artery (outflow of the aneurysm) was catheterized using a 2.8 Fr Progreat microcatheter (Terumo, Leuven, Belgium) (Fig. 1C). A 4 Fr Cobra catheter was placed inside the aneurysm via the right femoral artery. The lateral part of the aneurysm was then embolized via the 4 Fr Cobra using 23 0.035 MReye stainless steel coils with diameter ranging from 2 to 5 mm (Cook, Denmark) (Fig. 2A). Next, the aneurysm outflow and therefore several arteriovenous fistulas were embolized via the microcatheter using a Glubran2
123
L. Bonne et al.: Embolization of a Giant Intercostal Aneurysm and Arteriovenous Malformation
Fig. 1 A–C Selective angiogram of the fourth right intercostal artery in early arterial (A) and late arterial (B) phase revealing a giant postostial aneurysm connected with a pleural arteriovenous malformation.
Superselective angiogram of the aneurysm (C) shows abnormal arteries draining into pulmonary veins
Fig. 2 A–B Angiogram after coil embolization of the lateral and middle part of the aneurysm (A). The medial part is still patent and is now small enough to be embolized with glue via the microcatheter.
Control angiogram of the thoracic aorta (B) shows occlusion of the aneurysm and residual opacification of the cranial part of the AVM
123
L. Bonne et al.: Embolization of a Giant Intercostal Aneurysm and Arteriovenous Malformation
Fig. 3 A–C Control angiogram of the thoracic aorta after 6 months shows occlusion of the aneurysm (with coil mass) and residual opacification of the AVM
(GEM, Italy)/Lipiodol (Guerbet, France) mixture (1:3) under fluoroscopic control. The 4 Fr Cobra catheter was then removed. Further Glubran2/Lipiodol embolization of the aneurysm was performed while slowly pulling back the microcatheter, until the aneurysm inflow and the proximal part of the intercostal artery were closed. Subsequent angiograms of the thoracic aorta and the nearby intercostal arteries showed occlusion of the aneurysm and part of the pleural arteriovenous malformation (Fig. 2B). There were no complications. The patient was discharged from the hospital 3 days later. A 6-month control angiography of the thoracic aorta confirmed the occlusion of the intercostal aneurysm. Part of the pleural AVM was still supplied by other intercostal and thoracic arteries (Fig. 3).
Discussion Intercostal artery aneurysms and AVM have been described, but are rare. Most reported intercostal AVM are
thought to be secondary to trauma, interventional procedures such as biopsy or transthoracic surgery or inflammatory pleural disease. Only a couple of cases were treated using an exclusively endovascular approach. In this case report, we describe a giant intercostal artery aneurysm associated with a pleural arteriovenous malformation in a young patient who had suffered from bilateral spontaneous pneumothoraces in the previous years. Thoracic drainage of these pneumothoraces is thought to be the cause of development of the lesion, although the etiology of the giant aneurysm is not entirely clear. A primary traumatic cause is possible. Its origin, however, might be flow-related as well, analogous to the flow-related aneurysms seen in intracranial AVM. To our knowledge, this is the first case of a giant aneurysm of the intercostal artery associated with a diffuse pleural arteriovenous malformation. For this reason, and because of the general rarity of these lesions, this case was considered a therapeutic challenge. The aim of the endovascular procedure was filling the aneurysm with coils, occluding the outflow and inflow of
123
L. Bonne et al.: Embolization of a Giant Intercostal Aneurysm and Arteriovenous Malformation
the aneurysm with diluted glue while also embolizing the part of the pleural AVM supplied by the intercostal artery. We combined coil and glue embolization to increase the maximum occlusive effect and to avoid retrograde vascularization via arteries branching from the aneurysmal sac. Therefore, two catheter systems were placed, one inside the aneurysm and one in the outflow. By injecting diluted glue in the outflow, we occluded parts of the pleural AVM. After the aneurysm was heavily reduced by the coil mass, the outflow, the residual aneurysmal sac and the inflow of the intercostal artery could be occluded by slow injection of glue through the microcatheter during its withdrawal. In this way, we sealed the aneurysm in the long term, as was confirmed on a control angiography after 6 months. We instated this waiting time of 6 months for control angiography to allow potentially missed or occult feeders to hypertrophy and become angiographically detectable, as well as to allow coil compaction to occur. The residual pleural AVM was not treated because it is at the time asymptomatic. Moreover, the extension of the pleural AVM and the multiplicity of the feeders would be a real endovascular challenge. It is important to emphasize that in case of a lesion involving the descending thoracic aorta and the intercostal arteries, spinal vascularization should be visualized before proceeding to any therapeutic procedure, in order to prevent unintended spinal cord ischemia. The intercostal arteries give rise to several vessels that supply the spinal cord, with the dominant thoracic anterior radiculary or ‘Adamkiewicz’ artery mostly originating on the left side at level T9–T12. In our case, diagnostic angiography did not reveal any spinal arteries arising from the pathological intercostal artery, and since the Adamkiewicz artery
123
originated from an intercostobronchial artery on the left side, endovascular treatment seemed safe and appropriate. Conflict of interest of interest.
The authors declare that they have no conflict
References 1. Rivera PP, Kole MK, Pelz DM et al (2006) Congenital intercostal arteriovenous malformation. AJR 187:W503–W506 2. Coulter TD, Maurer JR, Miller MT et al (1999) Chest wall arteriovenous fistula: an unusual complication after chest tube placement. Ann Thorac Surg 67:849–850 3. Derdeyn CP, Middleton WD, Allen BT, Nordlicht SM (1993) Acquired intercostal arteriovenous fistula: color Doppler ultrasonographic diagnosis. J Ultrasound Med 12:679–681 4. Lai JH, Yan HC, Kao SJ et al (1990) Intercostal arteriovenous fistula due to pleural biopsy. Thorax 45:976–978 5. Siddhartha W, Parmar H, Shrivastav M et al (2000) Endovascular glue embolization of an intercostal arteriovenous fistula: a nonsurgical option. J Postgrad Med 46:213–214 6. Yamasaki N, Hata H, Kusaga M, Kubo K (1977) A surgical case of congenital intercostal arteriovenous fistula. Nippon Kyobu Geka Gakkai Zasshi 25:936–939 7. Saito A, Takahashi T, Ezura M, Tominaga T (2007) Intercostal arteriovenous fistula associated with neurofibromatosis manifesting as congestive myelopathy: case report. Neurosurgery 61:E656–E657 8. Hamilton MCK, Occleshaw CJ, Calder AL (2005) Congenital arteriovenous fistula between an intercostal artery and the left brachiocephalic vein. Cardiol Young 15:437–438 9. Gonzalez R, Santolaya R, Prats R, Rodriguez P (2010) Left intercostal aneurysm fistulised to azygos vein as a late complication of thoracic trauma. Eur J Cardiothorac Surg 39:276 10. Kim HJ, Seon HJ, Choi S, Jang NK (2011) Ruptured aneurysm of intercostal artery aneurysm associated with Neurofibromatosis Type 1: a case report. Cardiovasc Interv Radiol 34:S74–S77
