Case Report
Percutaneous Treatment of Bronchial Artery Aneurysm with Use of Transcatheter Coil Embolization and Thoracic Aortic Stent-Graft placement1 Toyohiko Sakai, MD Mahmood K. Razavi, MD Charles P. Semba, MD Stephen T. Kee, MD Daniel Y. Sze, MD, PhD Michael D. Dake, MD
Index terms: Aneurysm, therapy Artery, bronchial
JVIR 1998; 9:1025-1028
BRONCHIAL artery aneurysm is a rare condition, but when i t ruptures, it can cause a life-threatening hemorrhage. While surgery with aneurysm resection is considered the traditional therapy, there are several reports of endovascular treatment of this disorder with transcatheter arterial embolization (1-3). Recently, we evaluated a patient with a bronchial artery aneurysm originating a t the ostium of the vessel adjacent to the descending aorta that was successfully treated percutaneously with use of the combination of transarterial coil embolization and thoracic aortic stent-graft placement.
I CASE REPORT
From the Division of CardiovascularInterventional Radiology, H-3647, Stanford University Medical Center, Stanford University School of Medicine, 300 Pasteur Dr., Stanford, CA 94305. Received January 8, 1998; revision requested February 9; revision received and accepted March 20. Address correspondence to M.D.D. O SCVIR, 1998
A 72-year-old man was admitted to our hospital for further investigation and possible treatment of a suspected bronchial artery aneurysm that was incidentally found on a previous chest computed tomography (CT) examination to evaluate his chronic pulmonary disease. As a complication of a severe pulmonary infection during childhood, he developed diffuse cystic bronchiectasis that more severely affected his left lung. This was associated with chronic recurrent bronchopulmonary infections and two episodes of minimal hemoptysis in the last 18 months. At the time of his admission, he had no acute symptoms. On the CT scan of his chest, there was a round, 2.5-cm-diameter mediastinal mass adjacent to the descending aorta. It homogeneously enhanced after administration of intravenous contrast media and had the same attenuation as that of the aorta (Fig la). Small nodular and tubular structures were identified around the mass lesion, which also showed marked en-
hancement with contrast media and, therefore. were considered to be dilated and tortuous bronchial arteries (Fig lb). He was not a candidate for thoracotomy because of his advanced age and extremely poor respiratory function. Thoracic arteriography was performed to confirm the diagnosis of a bronchial artery aneurysm and to facilitate the planning of a n endovascular treatment. Thoracic aortography demonstrated a saccular aneurysm. The neck of the aneurysm was located a t the most proximal aspect of an enlarged tortuous left bronchial artery, very close to its origin from the descending aorta (Fig 2a). A selective left bronchial arteriogram identified two major outflow branches from the aneurysm (Fig 2b). Subsequently, the two outflow branches of the bronchial artery were selectively catheterized with use of a 3-F microcatheter (Tracker-18; Target Therapeutics, Fremont, CA) coaxially introduced through a 5-F Simmons I catheter (Cook, Bloomington, IN) placed a t the origin of the left bronchial artery. Both of these vessels were successfully embolized with 0.018-inch microcoils (one 5-mm Tornado coil [Cook] and three 7-mm coils, two 6-mm coils, two 2-mm coils [Target Therapeutics]). After the coil embolization, selective bronchial arteriography confirmed complete occlusion of the two outflow branches (Fig 2c). Subsequent thoracic aortography identified a small amount of residual flow of contrast media into the aneurysm (Fig 2d). Embolization of the origin of the bronchial artery, essentially within the wall of the aorta, was not a t t e m ~ t e dbecause it aDpeared impossible to precisely target the extremely short neck without sig-
1026
Percutaneous Treatment of Bronchial Artery Aneurysm
November-December 1998 JVIR
thrombosis (Fig 5). The patient was discharged from the hospital the same day. A follow-up CT scan obtained 7 months after stent-graft deployment demonstrated complete resolution of the aneurysm. I
DISCUSSION
,
"7A - % ---.---.
a.
1 h.
Figure 1. Chest CT images with contrast enhancement demonstrate (a)a round, well-enhanced mass (arrow) and (b) small nodular and ductal structures (arrow) adjacent to the descending aorta (a is the adjacent image caudal to b).
nificant risk of coil protrusion or frank embolization into the aorta. Therefore, stent-graft placement in the thoracic aorta was considered to completely exclude the blood flow into the aneurysm. The procedure was approved by the Institutional Review Board and informed consent was obtained from the patient. Based on measurements obtained from thoracic CT and aortography and a strategy of using a 10%-12% oversize for the neck diameter, the stent-graft was designed with a tapered configuration. The 5-cm-long device has a proximal diameter of 34 mm and distal diameter of 24 mm. The custom fabricated stent-graft consists of Z stents (Cook) of various diameters interconnected with use of 2-0 polypropylene suture and covered with 10-mm thinwall polytetrafluoroethylene (PTFE) graft material (Impra, Tempe, AZ) predilated with balloons of various diameters to form a tapered configuration that mirrors the underlying stent framework. The graft material is secured to the underlying stent with use of 6-0 polypropylene suture (Fig 3). The stent-graft procedure was per-
formed in the angiography suite the day after coil embolization. Thoracic aortography demonstrated persistent filling of the aneurysm with a small amount of contrast media. A 20-F sheath (Keller-Timmermans; Cook) was advanced from the right groin to the descending thoracic aorta over a stiff, 0.035-inch guide wire (Amplatz Super Stiff; Medi-techBoston Scientific, Watertown, MA). The sheath was positioned across the aneurysm and the tapered PTFE-covered stentgraft was deployed in the descending thoracic aorta, covering the origin of the bronchial artery supplying the aneurysm. Subsequently, thoracic aortography after stent-graft placement showed complete isolation of the bronchial artery aneurysm from the circulation and no leakage around the stent-graft (Fig 4). After the procedure, the patient was transferred to the operating room, the 20-F sheath was removed, and the artery was surgically repaired. A follow-up CT scan obtained the next day with intravenous administration of contrast media showed no enhancement of the bronchial artery aneurysm, consistent with complete
Bronchial artery aneurysm is commonly classified as either mediastinal or intrapulmonary according to its location. Mediastinal bronchial artery aneurysms are mostly asymptomatic, but there is a report of their rupture causing hemomediastinum, hematemesis, or symptoms mimicking dissecting aortic aneurysms (4). At the time of diagnosis, intrapulmonary bronchial artery aneurysms are usually associated with hemoptysis (2,5). Little is known about the etiology of bronchial artery aneurysms. There are several reports that suggest the pathophysiology of bronchial artery aneurysm is related to the hyperemia and arterial dilatation caused by persistent pulmonary parenchymal in. flammation, including bronchiectasis and tuberculosis (2,4). In the case presented, the patient had a long history of bronchiectasis. There are other reports describing bronchial artery aneurysm associated with a systemic vascular abnormality, such as OslerWeber-Rendu syndrome ( I ) and mycotic aneurysm of the bronchial artery complicating septicemia (3). Because bronchial artery aneurysm has a risk of fatal rupture, its treatment is considered whenever it is diagnosed, regardless of the presence or absence of symptoms (1,2,4,6,7). Open surgical treatment may include lobectomy, pneumonectomy, or ligation of the bronchial artery (2,6). Percutaneous endovascular treatment for bronchial artery aneurysm is an alternative therapy that is potentially less invasive and avoids surgical thoracotomy. In cases with massive hemoptysis, there are several reports describing the successful management of bronchial artery aneurysm with percutaneous transcatheter embolization (1-3). In a case reminiscent of ours, however, Remy-Jardin et a1 reported a procedure failure after they abandoned embolization of a bronchial artery aneurysm because the origin of the aneurysm was too close to the aorta to safely embolize (7). Endolu-
Sakai et a1
1027
Volume 9 Number 6
Figure 3. The stent-graft used in this case is 5 cm long and tapered from 34 mm to 24 mm in diameter. It consists of Z stents of varying diameter sutured together to form a taper and is covered with a segment of balloon-dilated PTFE graft material that mirrors the taper of the stent endoskeleton.
Figure 4. Descending thoracic aortography" after stent-graft placement dem-onstrates thrombosis of the aneurysm. A
Figure 2. (a)Descending thoracic aortography demonstrates a saccular aneurysm. The neck of the aneurysm is located at the most proximal aspect of the enlarged tortuous left bro~lchialartery. (b)Selective left bronchial arteriogaphy identifies two major outflow branches (arrows) from the aneurysm. ( c ) Selective left bronchial arteriography after coil embolization confirms complete occlusion of the two outflow branches. (d) Descending thoracic aortography demonstrates residual filling of contrast media into the aneurysm (arrow).
minal packing of the aneurysm itself, rather than its neck or feeding artery, with detachable balloons or steel coils is another alternative approach for such a case. However, based on published experiences of endovascular therapy for intracranial aneurysms,
1028
Percutaneous Treatment of Bronchial Artery Aneurysm
November-December 1998 JVIR
Figure 5. Image at the same level as in Figure l a from follow-up contrast material-enhanced chest CT performed the day after stent-graft placement demonstrates no enhancement of the aneurysm, consistent with complete thrombosis.
there is a persistent risk of aneurysm rupture even after packing with detachable balloons, and packing with coils is not as easy or effective for the treatment of relatively large aneurysms, such as the one in our case (8). After coil embolization and occlusion of the two bronchial run-off branches, residual, albeit reduced, flow of contrast media into the aneurysm was observed during aortography. Because of the direct contact between the aneurysm and the thoracic aorta and persistent exposure of the aneurysm sac to arterial pressure, further isolation and thrombosis of the aneurysm was advisable. The potential for spontaneous thrombosis was considered unlikely and less desirable than physical exclusion of the
aneurysm neck by ligature, coils, or graft material. Recently, we investigated the use of endoluminal stentgrafts for the treatment of descending thoracic aorta aneurysm (9,lO). This technology enables us to isolate blood flow to the thoracic aortic aneurysm. Consequently, the stent-graft also effectively seals the orifice of thoracic aortic branch arteries covered by the device. In the present case, the origin of the bronchial artery supplying the aneurysm was successfully occluded with use of a stent-graft. In addition to the occlusion of the inflow to the aneurysm, the distal outflow routes from the aneurysm were also embolized with microcoils to prevent the retrograde filling into the aneurysm. We used PTFE graft material to cover the stent-graft instead of woven polyester because a taper of 10 mm was necessary between the proximal and distal ends of the stent-graft, and polyester grafts with a gradual taper are not readily available. The diameter of a Z stent-based stent-graft depends on the diameters of the proximal and distal necks of the aneurysm. Based on our thoracic stent-graft experience, tapering of a stent-graft is necessary when there is a difference of greater than 10% between neck diameters. A report detailing the stentgraft treatment of iliac artery aneurysm with a Z-stent device recommends the use of a tapered stent-graft covered with PTFE when the difference in diameter between the proximal and distal necks exceeds 2 mm (11). In the present case, we successfully isolated a large bronchial artery aneurysm from the circulation with a combination of thoracic aortic stentgraft occlusion of inflow and coil embolization of outflow branches. Longterm follow-up of this case and further experiences of similar cases are necessary, however, to prove the true feasibility and advisability of this technique given the potential risks of perigraft leak, deterioration or migration of the stent-graft, or retrograde
filling of the aneurysm resulting from the recanalization of the embolized bronchial artery. References 1. Ishizaki 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; 25:852-854. 2. Osada H, Kawada T, Ashida H, Sodemot0 U, Noguchi T. Bronchial artery aneurysm. Ann Thorac Surg 1986; 41: 440-442. 3. Servoice V, Denys A, Sibert A. Mycotic aneurysm of the bronchial artery: a rare cause of hemoptysis. AJR 1992; 159:428. 4. Hall RCJ, Miller GAH, Keller IH. Ruptured bronchial artery aneurysm mimicking aortic dissection. Br Heart J 1977; 39:909-910. 5. Shear AH, Bashist B. Computed tomography of bronchial artery aneurysm with erosion into esophagus. J Comput Assist Tomogr 1989; 13:1069-1071. 6. Hoffman V, Ysebaert D, De Sphepper A, Colpaert C, Jorens P. Acute superior vena cava obstruction after rupture of a bronchial artery aneurysm. Chest 1996; 110:1356-1358. 7. Remy-Jardin M, Remy J , Ramon PH, Fellous G. Mediastinal bronchial artery aneurysm: dynamic computed tomography appearance. Cardiovasc Intervent Radiol 1991; 14:118-120. 8. Byrne J. Review article: endovascular treatments for intracranial aneurysms. Br J Radiol 1996; 69:891-899. 9. Dake MD, Miller DC, Semba CP, Mitchell RS, Walker PJ, Liddell RP. Transluminal placement of endovascular stent-grafts for the treatment of descending thoracic aortic aneurysm. N Engl J Med 1994; 331:17291734. 10. Fann JI, Dake MD, Semba CP, Liddell RP, Pfeffer TA, Miller DC. Endovascular stent-grafting after arch aneurysm repair using "elephant trunk." Ann Thorac Surg 1995; 60:1102-1105. 11. Razavi MK, Dake MD, Semba CP, Nyman UO, Liddell RP. Percutaneous endoluminal placement of stent-grafts for the treatment of isolated iliac artery aneurysm. Radiology 1995; 197: 801-804.