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Late follow-up after transcatheter occlusion of a thoracic aortic aneurysm for a premature infant John F. Rhodes Jr.a,⁎, Jeffrey W. Delaneyb, James C. Fudge Jr.c a b c
Miami Children's Hospital, Miami, FL, United States Children's Omaha, University of Nebraska, United States University of Florida, Gainesville, FL, United States
1. Introduction Thoracic aortic aneurysms are uncommonly diagnosed in children [1] and thus there are few reports in the literature [2,3]. When children are found to have this medical condition there remains controversy as to medical versus surgical management. Our goal was to describe the technique of transcatheter occlusion of an aortic aneurysm in an infant with late follow-up regarding the outcome of this procedure.
and posterior. In addition, there was concern for mild-to-moderate focal narrowing of the adjacent descending aorta at the proximal margin of the aneurysm. At 5 months of age, the infant was referred to our medical center and discussed in our multidisciplinary conference. As a result of this discussion, the infant was referred to the pediatric cardiac catheterization laboratory for further angiographic evaluation of the aneurysm, assessment of the aortic narrowing, and possible transcatheter occlusion.
2. Case Report
2.1. Transcatheter Technique
A 5 month old, born at 26-weeks gestation, was referred to our institution for management of a descending thoracic aortic aneurysm which was discovered during a prolonged neonatal hospitalization. The infant weighed 890 g at birth and was born via emergent cesarean section secondary to prolonged premature rupture of membranes and suspected chorioamnionitis. At birth, the infant required intubation and mechanical ventilation for respiratory distress syndrome, as well as, placement of an umbilical artery catheter for hemodynamic monitoring. Early in the hospital course, a patent ductus arteriosus was discovered and treated with indomethacin. Subsequently, the infant developed Staphylococcus aureus sepsis requiring a three-week treatment with Naficillin, Gentamicin, and Rifampin. Follow up echocardiography confirmed closure of the ductus arteriosus but revealed an echolucent area adjacent to the descending thoracic aorta which was concerning for an aortic aneurysm. Subsequent echocardiograms revealed an increase in size of this echo-lucent area; therefore, magnetic resonance imaging (MRI) was performed. Serial MRI was non-diagnostic and revealed minimal change in the size of the aneurysm. At 3 months of age, the infant was taken to the operating room for thoracic exploration and the descending thoracic aortic aneurysm was identified but not repaired as it was deemed necessary to refer the neonate to a center with cardiovascular surgery. At 4 months of age, a computed tomography (CT) angiogram revealed a saccular descending thoracic aortic aneurysm at the level of the seventh and eighth thoracic spine (Fig. 1). The aneurysm measured 9 mm × 13.5 mm × 11.8 mm and there was a moderate mass effect with deviation of the aorta leftward
Hemodynamic measurements and angiography were performed using a 4 French end-hole catheter and a 4 French pigtail catheter, respectively. There was no hemodynamic evidence of aortic obstruction. Initial cineangiograms were obtained in the descending aorta just above the diaphragm in the right anterior oblique (20°) and left anterior oblique (70°) views. The angiogram demonstrated a saccular aneurysm (10 mm × 12 mm) with a narrow stalk (3.9 mm) arising from the right side of the descending thoracic aorta (Fig. 2). Although there was mass effect of the aneurysm resulting in leftward compression of the descending aorta and moderate narrowing (3.2 mm) of the aorta just proximal to the aneurysm there was unobstructed flow of contrast from the descending thoracic aorta to the intra-abdominal aorta. Also, there was normal branching of the celiac and superior mesenteric arteries off the abdominal aorta. The normal descending thoracic aorta proximal to the level of the aneurysm measured 5.5 mm. Subsequently, the femoral arterial sheath was exchanged for a 6 French sheath and a 6 French MPA-1 guide catheter (Cordis, Bridgewater, NJ) was modified by shorting the length and placing a 5 French sheath valve on the back of the guide. This modified catheter was then advanced to the thoracic aorta and a 4F angle glide catheter (Terumo, Somerset, NJ) was coaxially placed through the modified MPA-1. Using the glide catheter, a 0.35″ J wire was positioned in the body of the aneurysm and the modified MPA-1 guide catheter was then advanced over the wire and angle glide into the aneurysm. The 4F wire and angle glide were removed. Initial attempts were made at placement of a 4 mm Amplatzer vascular plug (AVP) (St. Jude, Minneapolis) into
⁎
Corresponding author at: Miami Children's Health System, 3100 SW 62 Avenue, Miami, FL 33155-3009, United States. E-mail address:
[email protected] (J.F. Rhodes).
http://dx.doi.org/10.1016/j.ppedcard.2017.05.003 Received 6 September 2016; Received in revised form 17 March 2017; Accepted 1 May 2017 1058-9813/ © 2017 Published by Elsevier Ireland Ltd.
Please cite this article as: Rhodes, J.F., Progress in Pediatric Cardiology (2017), http://dx.doi.org/10.1016/j.ppedcard.2017.05.003
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Fig. 3. A 10 mm Amplatzer vascular plug deployed across the aneurysm stalk with protrusion into the aorta.
Fig. 1. Computed tomography angiogram reveals a saccular descending thoracic aortic aneurysm at the level of the seventh and eighth thoracic spine.
Fig. 4. Descending aorta angiography demonstrates the 10 mm Amplatzer vascular plug in the aortic aneurysm with near complete occlusion and no change in the mass effect on the aorta.
Fig. 2. Direct angiography of the aorta demonstrates a 10 mm × 12 mm saccular aneurysm with a 3.9 mm stalk arising from the right side of the descending thoracic aorta. There is mass effect of the aneurysm resulting in leftward compression of the descending aorta and moderate narrowing of the aorta just proximal to the aneurysm. The normal descending thoracic aorta proximal to the level of the aneurysm measured 5.5 mm.
aorta with aortic narrowing that was consistent with the pre-intervention angiogram (Fig. 4). The patient was extubated and received prophylactic antibiotics for 24 h prior to discharge the following day. An echocardiogram performed on the day of discharge showed the AVP in stable position with unobstructed flow through the aorta.
the neck of the aneurysm via the modified guide catheter; however, the AVP was not deployed as the diameter of the plug resulted in an unstable position. Therefore, a 10 mm AVP (St. Jude, Minneapolis) was advanced into the neck of the saccular aneurysm via the modified MPA1 catheter. The 10 mm AVP was positioned so that the majority of the plug was within the body of the aneurysm, with a small portion protruding into the aortic lumen. The AVP was then deployed and an angiogram was performed revealing narrowing of the aorta at the site of the protrusion of the AVP (Fig. 3). Due to a 30–40 mm Hg gradient with the AVP in this position the plug was pushed within the saccular aneurysm with no further protrusion into the thoracic aorta. After the AVP was repositioned, there was no further evidence of anatomic or hemodynamic obstruction of the descending aorta. Hemodynamic measurements revealed a proximal descending thoracic aortic pressure, 85/52 mm Hg compared to the descending abdominal aortic pressure of 82/52 mm Hg. Post-intervention angiograms demonstrated free flow of contrast from the descending thoracic to the descending abdominal
2.2. Late Follow-Up The patient was followed yearly since the procedure with normal growth and development. The patient is now 10 years of age has had no evidence of aortic obstruction by physical exam, upper and lower extremity blood pressures, or transthoracic echocardiography. At the latest follow up visit the echocardiogram showed the device to be stable with no demonstrable flow into the aneurysm and no evidence of aortic compression, coarctation, or new aneurysmal formation. In addition, a CT angiogram at five-year follow-up demonstrated complete occlusion of the aneurysm with the AVP in a good position and no new aneurysmal formation (Fig. 5). The mass effect from the AVP occluded aneurysm was unchanged. 2
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aortic aneurysm should be considered as a management option. For an infant and consequently small descending aorta the AVP can be placed directly within the saccular aneurysm with complete closure and no clinically significant distortion of the aorta at late follow-up. 4. Discussion Although thoracic aortic aneurysms are uncommon in infants and children they can often be associated with morbidity and mortality [1–3]. They are also often difficult to identify and should be considered for anyone with a history of central arterial catheters [3–6] or after extracorporeal cannulation [7]. Our case demonstrates that the aneurysm can have a compression effect on the thoracic aorta but not have a significant gradient while sedated in the catheterization laboratory. The concern is a dynamic effect, late thrombosis with embolic events, as well as progressive enlargement over weeks to years. Although surgical management has been utilized historically for this vascular condition (3–4) our patient demonstrates with late follow up that complete occlusion of the aneurysm and no residual aortic obstruction can be accomplished by closure using a percutaneous vascular device within the saccular aneurysm. References [1] Sarkar R, Coran AG, Cilley RE, et al. Arterial aneurysms in children: clinicopathologic classification. J Vasc Surg 1991;13:47–56. [2] Wyers MR, McAlister WH. Umbilical artery catheter use complicated by pseudoaneurysm of the aorta. Pediatr Radiol 2002;32:199–201. [3] Mendeloff J, Stallion A, Hutton M, et al. Aortic aneurysm resulting from umbilical artery catheterization: case report, literature review, and management algorithm. J Vasc Surg 2001;33:419–24. [4] Lobe TE, Richardson CJ, Boulden TF, et al. Mycotic thrombo aneurysmal disease of the abdominal aorta in preterm infants: its natural history and its management. J Pediatr Surg 1992;27:1054–9. [discussion 1059–60]. [5] Blondiaux E, Miquel J, Thomas P, et al. Calcified aneurysm of the abdominal aorta 12 years after umbilical artery catheterization. Pediatr Radiol 2008;38:233–6. [6] Adelman RD. Abdominal aortic aneurysm 18 years after apparent resolution of an umbilical catheter-associated aortic thrombosis. J Pediatr 1998;132:874–5. [7] Paul JJ, Desai H, Baumgart S, Wolfson P, Russo P, Tighe DA. Aortic dissection in a neonate associated with arterial cannulation for extracorporeal life support. ASAIO J 1997;43:92–4.
Fig. 5. Five year follow-up CT angiogram demonstrates complete occlusion of the aneurysm with a good position of the Amplatzer vascular plug as well as no new aneurysmal formation. The mass effect from the Amplatzer vascular plug occluded aneurysm was unchanged.
3. Conclusion This report demonstrates that transcatheter occlusion of a thoracic
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