Surgical carotid pseudoaneurysm repair following percutaneous catheterization: a case report

Introduction

Pulmonary atresia with intact ventricular septum (PA-IVS) is a rare congenital heart disease (CHD) where the pulmonary valve and right ventricle do not properly form. The majority of patients with PA-IVS undergo systemic-to-pulmonary shunt placements such as a modified Blalock-Taussig-Thomas (mBTT) shunt or ductal stents as stage 1 palliation (1). Approximately 7% of mBTT shunts malfunction within 30-day post-operation (2). Interventional shunt stenting can be an effective treatment to restore patency of occluded or stenotic mBTT shunts (3,4). For infant patients, the utilization of percutaneous carotid access (PCA) is gaining in popularity (5). When compared to femoral access, PCA enables a more direct route to the aortic arch through a larger artery (6).

A rare complication of PCA in the infant population is the formation of a pseudoaneurysm (6). Given the rarity of this complication the optimal methods to screen for, diagnose, and manage carotid pseudoaneurysms in the infant population is not clearly defined.

We describe the case of an infant with PA-IVS who underwent PCA catheterization to stent an occluded mBTT shunt and later developed a common carotid artery pseudoaneurysm. The pseudoaneurysm was successfully managed surgically. We present this case in accordance with the CARE reporting checklist (available at https://asj.amegroups.com/article/view/10.21037/asj-24-22/rc).

Case presentation

A newborn male with prenatally diagnosed PA-IVS was born at 34-week gestation via induced vaginal delivery. The baby’s birth weight was 1.8 kg. Apgar scores were 8 and 9 at 1- and 5-minute respectively. Following birth, the patient was admitted to the neonatal intensive care unit (NICU). While in the NICU, that patient received prostaglandin E2 to maintain a patent ductus arteriosus (PDA). The patient required bubble continuous positive airway pressure (CPAP) and achieved an oxygen saturation of ~85%.

At 55 days old, 3.3 kg, the patient underwent surgery (Table 1). While the patient’s coronary blood supply was not dependent on the right ventricle, significant right ventricle outflow tract (RVOT) submuscular crowding made an interventional pulmonary repair not feasible. The patient underwent an RVOT augmentation, right ventricular (RV) muscle bundle resection, PDA ligation, right atrium (RA) reduction plasty, and placement of a 3.5-mm Gore-Tex graft (W.L. Gore & Associates, Flagstaff, AZ, USA) mBTT shunt to the main pulmonary artery. Post-operation, the patient was transferred to the pediatric cardiac intensive care unit (PCICU) where he was maintained on heparin 15 units/kg/h for shunt thrombosis prophylaxis.

On post-operative-day (POD) 10, 65 days old, the patient was found to have desaturations and an echocardiogram showed complete mBTT shunt thrombosis. Two bolus doses of 100 units/kg of heparin were administered in addition to the continuous heparin drip. The child was taken to the catheterization lab emergently. Percutaneous right common carotid artery access was obtained utilizing a 4-French sheath. A drug releasing Synergy XD (Boston Scientific, Marlborough, MA, USA) 3.5 mm by 32 mm stent was placed covering the entire length of the mBTT shunt and restoring patency (Figure 1). Post-intervention, the patient’s oxygen saturation improved from ~60% to ~80%. Upon sheath removal, hemostasis was achieved with manual pressure.

Figure 1 Catheterization images: (A) occluded mBTT shunt (red arrow), (B) patent mBTT shunt post stent deployment (white arrow). Note the sheath is in the carotid artery. mBTT, modified Blalock-Taussig-Thomas.

Following the catheterization, the patient’s heparin dose was increased from 15 to 30 units/kg/h. On POD-19, 74 days old, the patient was transitioned to enoxaparin 5 mg every 12 h, clopidogrel 0.2 mg/kg daily, and aspirin 20.25 mg daily.

Over the subsequent days, the patient remained in the PCICU gaining weight and being weaned off respiratory support while continuing treatment with enoxaparin, clopidogrel, and aspirin. On POD-39, 94 days old, a non-tender nodule was noted on the patient’s right neck during physical exam. Color doppler ultrasound revealed a mass medial to the carotid artery with to-and-fro flow with arterial waveforms highly suspicious for a pseudoaneurysm (Figure 2). Computed tomography (CT) angiogram with contrast confirmed the pseudoaneurysm measuring 1.8 cm × 1.7 cm × 2.1 cm arising from the common carotid artery (Figure 3). As interventional repairs were not feasible, our multidisciplinary team including pediatric cardiothoracic surgery, vascular surgery, pediatric interventional cardiology, and pediatric intensivists decided to treat the pseudoaneurysm surgically.

Figure 2 Preoperative ultrasounds: (A) right carotid pseudoaneurysm (red arrow), (B) right carotid pseudoaneurysm with to-and-fro flow (white arrow), (C) communication between the carotid artery and the pseudoaneurysm (yellow star). JUG, jugular; TRV, transverse; RT, right; LAT, lateral; INF, inferior.

Figure 3 Preoperative computed tomography showing a right carotid pseudoaneurysm. Blue (1.8 cm) and yellow (1.7 cm) markers measure the size of the pseudoaneurysm.

On POD-48, 103 days old, the patient underwent surgical pseudoaneurysm repair. An oblique incision was made medial to the sternocleidomastoid muscle and the right carotid artery was isolated superior to the suprasternal notch. Proximal control of the right carotid artery was obtained (Figure 4A) and the pseudoaneurysm sac carefully dissected. Distal control of the carotid was then obtained, the carotid was snared at the distal and proximal control points, and the sac was opened and excised. There was a small communication between the pseudoaneurysm and right carotid artery (Figure 4B) which was closed utilizing 5-0 Prolene sutures in a continuous manner. The carotid snares were released, hemostasis achieved, and functional carotid pulses observed (Figure 4C). Figure 5 shows the excised pseudoaneurysm sac. The patient was extubated in the operating room. Post-operative ultrasound demonstrated no pseudoaneurysm and a patent carotid artery with normal waveforms (Figure 6).

Figure 4 Intraoperative photos: (A) the carotid pseudoaneurysm, the retracted sternocleidomastoid muscle and proximal control of the carotid artery; (B) opened pseudoaneurysm sac showing the point of communication; (C) carotid artery after repair and excision of the pseudoaneurysm.

Figure 5 Excised pseudoaneurysm sac: (A) intraluminal surface, (B) extraluminal surface.

Figure 6 Postoperative ultrasound showing a successful pseudoaneurysm repair and patent right common carotid artery (red arrows): (A) sagittal view, (B) transverse view.

The remaining post-operative course was uneventful. The patient was discharged home 14 days later at 117 days old. While the patient required additional cardiac surgeries, there were no complications associated with the carotid pseudoaneurysm repair. At the most recent follow-up, 2-years post-operation, the patient was growing well and meeting his development milestones (e.g., normal Bayley’s cognitive scale composite score and 47^th percentile stature-for-age) with no signs of neurological sequalae from the carotid artery repair.

All procedures performed in this study were in accordance with the ethical standards of the institutional research committee and with the Helsinki Declaration (as revised in 2013). Written informed consent was obtained for the publication of this case report and accompanying images from the patient’s guardians. A copy of the written consent is available for review by the editorial office of this journal.

Discussion

We describe a carotid artery pseudoaneurysm that developed 29 days following PCA intervention to place a stent in an occluded mBTT shunt. The carotid pseudoaneurysm was successfully repaired with surgery. The development of a pseudoaneurysm 29 days post intervention is later than other reported cases.

The strengths of our case report include the detailed clinical history, intra-operative photos, and a 2-year follow-up. An inherent limitation of our case report is that it is a single data-point and therefore lacks the statistical rigor to draw definitive conclusions or make definitive recommendations.

Choudhry et al. report a case series of 20 PCA interventions in infants <3 months from 2012–2015, with two patients developing carotid pseudoaneurysms within 24 h. Both patients were successfully treated surgically (6). In a case series of 61 pediatric PCA catheterizations, Ligon et al. report no pseudoaneurysms and Justino et al. (n=47) report 1 pseudoaneurysm, which required surgical repair (3,7). In the most recent case series of 45 procedures from 2020–2022, median age 15 days, six clinically silent pseudoaneurysms were detected on ultrasound at 1 or 7 days post-procedure all of which spontaneously resolved (5).

Our patient’s significant anticoagulation and antiplatelet exposure (i.e., 19 days of heparin treatment and 20 days receiving enoxaparin, clopidogrel, and aspirin) before and after the catheterization increased the risk of post-PCA pseudoaneurysm formation. An adult study analyzing risk factors for the development of femoral pseudoaneurysms following catheterization (n=401) found a positive association with post-catheterization anticoagulation [odds ratio (OR) =4.42, P<0.01] and periprocedural P2Y12 inhibitors (OR =2.95, P=0.02) (8). Reducing the anticoagulant and antiplatelet therapy could have lowered the risk of pseudoaneurysm formation, however, it would have also increased the risk of a recurrent thrombotic event.

Once the pseudoaneurysm was identified, multiple treatment options were evaluated. Given the large size of the aneurysm (1.8 cm × 1.7 cm × 2.1 cm), conservative management was deemed inappropriate given the high risk of rupture, continued enlargement, and compression of adjacent structures. While stents have been successfully utilized in adults, there are no approved dilatable stents for infants (9). We considered ultrasound-guided thrombin injections, a common treatment option for pediatric femoral pseudoaneurysms, however, we felt that the risk of a distal emboli and stroke was too high (10). Finally, coiling of the carotid artery pseudoaneurysm was deemed infeasible. Surgical repair was the selected treatment as it would provide a durable solution while minimizing the risk of serious complications (e.g., stroke) (11).

This case report provides a detailed history of a unique carotid pseudoaneurysm 29 days after PCA catheterization in an infant with significant anticoagulation exposure. This is of particular value to providers treating CHD as PCA gains in popularity. Additional research is needed to determine the optimal management strategy of post-PCA pseudoaneurysm repairs in infants. Providers should consider reducing the exposure to antiplatelets and anticoagulants, post-procedure, to decrease the risk of pseudoaneurysm formation. We must also have a high index of suspicion for the development of pseudoaneurysms when antiplatelets/anticoagulation cannot be weaned quickly and consider performing a surveillance ultrasound to detect such complications early. However, the timing of such surveillance is not clearly known.

Conclusions

We describe a carotid pseudoaneurysm that was identified 29 days after PCA catheterization and successfully treated with surgery. As PCA gains in popularity, providers should be aware of the risk of late developing pseudoaneurysm, especially in patients with significant anticoagulation exposure, have a high index of suspicion, and be prepared to quickly diagnose and appropriately manage the pseudoaneurysm to avoid any complications including rupture.

Acknowledgments

Funding: None.

Footnote

Reporting Checklist: The authors have completed the CARE reporting checklist. Available at https://asj.amegroups.com/article/view/10.21037/asj-24-22/rc

Peer Review File: Available at https://asj.amegroups.com/article/view/10.21037/asj-24-22/prf

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://asj.amegroups.com/article/view/10.21037/asj-24-22/coif). R.M. serves as an unpaid editorial board member of AME Surgical Journal from September 2023 to August 2025. The other authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration (as revised in 2013). Written informed consent was obtained for the publication of this case report and accompanying images from the patient’s guardians. A copy of the written consent is available for review by the editorial office of this journal.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

References

1. Cheung EW, Mastropietro CW, Flores S, et al. Procedural Outcomes of Pulmonary Atresia With Intact Ventricular Septum in Neonates: A Multicenter Study. Ann Thorac Surg 2023;115:1470-7. [Crossref] [PubMed]
2. Headrick AT, Qureshi AM, Ghanayem NS, et al. In-hospital Morbidity and Mortality After Modified Blalock-Taussig-Thomas Shunts. Ann Thorac Surg 2022;114:168-75. [Crossref] [PubMed]
3. Ligon RA, Kim DW, Vincent RN, et al. Angiographic follow-up of infants and children undergoing percutaneous carotid artery interventions. Catheter Cardiovasc Interv 2018;91:1301-6. [Crossref] [PubMed]
4. Ligon RA, Ooi YK, Kim DW, et al. Intervention on Surgical Systemic-to-Pulmonary Artery Shunts Carotid Versus Femoral Access. JACC: Cardiovascular Interventions 2017;10:1738-44. [Crossref] [PubMed]
5. Mejia E, McLennan D, Zablah J, et al. Establishing Carotid Seldinger as Routine Access in Infants; Planning, Performance, and Follow-Up Protocols. Pediatr Cardiol 2023;44:1815-20. [Crossref] [PubMed]
6. Choudhry S, Balzer D, Murphy J, et al. Percutaneous carotid artery access in infants < 3 months of age. Catheter Cardiovasc Interv 2016;87:757-61. [Crossref] [PubMed]
7. Justino H, Petit CJ. Percutaneous Common Carotid Artery Access for Pediatric Interventional Cardiac Catheterization. Circ Cardiovasc Interv 2016;9:e003003. [Crossref] [PubMed]
8. Naddaf A, Williams S, Hasanadka R, et al. Predictors of Groin Access Pseudoaneurysm Complication: A 10-Year Institutional Experience. Vasc Endovascular Surg 2020;54:42-6. [Crossref] [PubMed]
9. Berman DP, Morray B, Sullivan P, et al. Results of the multicenter early feasibility study (EFS) of the Renata Minima stent as treatment for branch pulmonary artery stenosis and coarctation of aorta in infants. Catheter Cardiovasc Interv 2024;104:61-70. [Crossref] [PubMed]
10. Boubalos JJ, Connolly BL, Amaral JG, et al. Ultrasound-Guided Thrombin Injection for the Treatment of Femoral Pseudoaneurysm in Pediatric Patients. J Vasc Interv Radiol 2016;27:519-23. [Crossref] [PubMed]
11. Kızılkaya MH, Biçer M, Ödemiş E, et al. A rare complication after an interventional procedure using the common carotid: carotid pseudoaneurysm in an infant. Cardiol Young 2023;33:1436-9. [Crossref] [PubMed]