Case Report. A 22-year-old female presented to an outside hospital with back pain radiating to the epigastrum. The patient had a history of hypertension and Takayasu’s arteritis. At age 17 she underwent resection of an ascending aortic aneurysm via a median sternotomy with placement of a graft to the aortic arch and banding of the sino-tubular junction because of significant aortic regurgitation. Six weeks prior to her presentation with back pain, the patient underwent resection of a descending thoracic aortic aneurysm via a left posterior-lateral thoracotomy and placement of a 26 mm graft. This included resection of a portion of the aortic arch (hemi-arch) to the descending thoracic aorta just cranial to the diaphragm.
The workup in this patient with back pain occurring 6 weeks post-operation included computed tomographic (CT) imaging with intravenous contrast. This demonstrated a pseudo-aneurysm arising from a focal defect of the mid portion of the recently placed descending thoracic aortic graft. The patient was hemodynamically stable and transferred to our institution on the same day she presented to the emergency room of the outside hospital.
Endovascular stent-graft repair was anticipated. Upon arrival to our institution, the patient was taken immediately to conventional invasive X-ray angiography, which confirmed that the pseudoaneurysm arose from a focal defect in the mid portion of the descending thoracic aortic graft at the level of the main pulmonary artery (Figure 1). The aortic graft in the transverse dimension measured 30 mm, which had increased in size from the 26 mm graft placed at surgery. The focal defect was amenable to endovascular stent-graft repair; however, angiography of the femoral and iliac vessels demonstrated a transverse size of 6.5 mm. In order to place the appropriate-sized endovascular stent-graft, at least a 20 Fr arterial sheath (diameter of 6.7 mm) would need to be used. However, this was too large a size to be safely placed in the patient’s peripheral vessels.
Because of the focal nature of the pseudo-aneurysm and the inability to place an endovascular stent-graft, the use of endovascular occluder devices was contemplated. With the consent of the patient’s family and institutional review board under compassionate use guidelines, an Amplatzer patent foramen ovale (PFO) occluder device was used to seal the connection between the aortic graft lumen and the focal pseudoaneurysm immediately after angiography was performed (Figure 2). A 5 Fr Judkins right (JR) 4 catheter was advanced into the pseudoaneurysm over a movable-core 0.035 inch wire. The JR 4 catheter was replaced with an 8 Fr transseptal sheath, then the distal limb of an 18 mm Amplatzer PFO occluder device was successfully deployed in the pseudo-aneurysm. After deployment of the distal and proximal limbs of the PFO device, it appeared to be flat against the aortic graft wall. Tugging on the device demonstrated adequate and stable placement. Transesophageal echocardiography was utilized for guidance during the procedure (Figure 3). Angiography after device placement demonstrated no evidence of contrast within the pseudo-aneurysm (Figure 2C).
On the following day after percutaneous closure of the pseudoaneurysm, the patient underwent a repeat CT with intravenous contrast, which demonstrated adequate placement of the device without contrast extravasation (Figures 4 and 5). The patient was discharged home the day following the procedure on daily aspirin therapy. With radiographic and clinical follow up at 34 months, the patient has done well and not required any additional procedures (Figure 6).
Discussion. This patient had a complication of Takayasu’s arteritis, progressive aortic aneurysmal disease. Medical therapy is the mainstay of treatment for patients with Takayasu’s arteritis and is aimed at halting the inflammatory process with the use of immune-modulating medicines.1 Surgical therapies are primarily aimed at the treatment of symptoms of end-organ ischemia due to critical stenoses with vessel bypass or preventing the risk of dissection/rupture due to aneurysmal disease by vascular reconstruction with graft material.1
The leading cause of surgical complications in patients with Takayasu’s who have undergone surgery is anastomotic aneurysms.2 The patient described in this report underwenttwo prior aortic reconstructive surgeries. Because of this, the initial concern was that this pseudo-aneurysm was likely secondary to the surgical anastomotic site where there may have been impaired healing between the graft and native aorta. Upon close inspection, the pseudo-aneurysm did not occur at the anastomotic site, but rather in the mid portion of the graft (Figure 5). This is a very uncommon finding. It is our hypothesis that the pseudo-aneurysm was caused by the intraoperative measurement of pressure within the graft by needle catheterization connected to fluid-filled tubing and expansion of the puncture site over time. This is not infrequently performedintraoperatively to ensure there is no increased intra-aortic pressure secondary to malposition of the graft. However, pseudo-aneurysms arising from needle catheterization sites are not common.
Open surgical repair was an option, but given this patient’s clinical course to date, it seemed probable that the patient would need future thoracic surgical repair. To minimize this, an endovascular approach using a stent-graft was considered a rational approach due to its likelihood of reduced morbidity and mortality. However, the patient’s peripheral vessel size for delivery of a stent-graft device was a limitation. An Amplatzer PFO occluder device was felt to be an acceptable device for several reasons: the device can be placed using an 8 Fr catheter (diameter of 2.7 mm); the design of the device was considered to allow for a favorable conformation to the arterial wall; and the narrow waist of the PFO occluder device (in comparison with the atrial septal occluder (ASD) device) was considered favorable in its design by limiting additional force to the graft material and potentially enlarging the hole. The ease of removability of the device was also favorable in the event the device did not perform as desired during implantation.
Percutaneous closure of vascular defects/structures is a therapeutic option with reduced mortality and morbidity. However, the risks and benefits must be individualized to each case and surgical consultation is strongly recommended. Stent-grafts are an option, but require larger vascular access sheaths. Other endovascular devices requiring smaller vascular access sheaths have been used for closure of a surgical graft anastomotic leak (Amplatzer ASD)3 or endoleaks from stent-graft placements (Amplazter vascular plug).4 Utilizing some of the devices may be an off-label indication, and operators and patients should be aware of this. This case report describes the successful use of a percutaneously placed Amplazter PFO occluder device to close an aortic pseudo-aneurysm arising from an aortic graft.
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2. Miyata T, Sato O, Koyama H, et al. Long-term survival after surgical treatment of patients with Takayatsu’s arteritis. Circulation 2003; 108: 1474– 1480.
3. Alzubaidi A, MacDonald S, Clement J, Webb JG. Percutaneous closure of a paraanastomotic abdominal aortic graft leak. Catheter Cardiovasc Interv 2006; 68: 799– 802.
4. Ferro C, Petrocelli F, Rossi UG, et al. Vascular percutaneous transcatheter embolisation with a new device: Amplatzer vascular plug. Radiol Med 2007; 112: 239– 251.