Percutaneous coronary intervention (PCI) is an extremely common and well-established procedure in adults which is rarely performed in children. We present a case of a successful left main coronary artery stenting in a small infant with a congenital coronary artery anomaly. We highlight the technical challenges of performing a PCI in a small patient, the risks of antithrombotic prophylaxis in this age group, and the importance of the combined work of the adult and pediatric interventional cardiologist.info:eu-repo/semantics/publishedVersio

Lourenço, G

Martins, JD

Pinto, MF

Sousa, L

English

Repositório do Centro Hospitalar de Lisboa Central, EPE

Case ReportA Rare but Real Necessity: Case Report of Coronary ArteryStenting in an InfantJosé D. Martins ,1 Guilherme Lourenço ,1 Lídia Sousa ,2 and Fátima F. Pinto 11Pediatric Cardiology Department, Hospital de Santa Marta, Centro Hospitalar de Lisboa Central EPE, Lisbon, Portugal2Cardiology Department, Hospital de Santa Marta, Centro Hospitalar de Lisboa Central EPE, Lisbon, PortugalCorrespondence should be addressed to José D. Martins; jdferreiramartins@gmail.comReceived 15 April 2021; Accepted 16 January 2022; Published 22 February 2022Academic Editor: Manabu ShirotaniCopyright © 2022 José D. Martins et al. This is an open access article distributed under the Creative Commons AttributionLicense, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work isproperly cited.Percutaneous coronary intervention (PCI) is an extremely common and well-established procedure in adults which is rarelyperformed in children. We present a case of a successful left main coronary artery stenting in a small infant with a congenitalcoronary artery anomaly. We highlight the technical challenges of performing a PCI in a small patient, the risks ofantithrombotic prophylaxis in this age group, and the importance of the combined work of the adult and pediatricinterventional cardiologist.1. IntroductionPercutaneous coronary intervention (PCI) is a very commonprocedure in adults, but extremely rare in children. In thelatter, coronary artery stenosis requiring intervention isexceptional, particularly in infancy, where we found onlyseven such cases [1–6]. Older children or adolescents mayalso present with coronary compromise requiring PCI, suchas after percutaneous pulmonary valve implantation [7],Kawasaki disease [8], or heart transplantation [9].Congenital coronary anomalies are sporadic findingsthat occur either as an isolated anomaly of the coronary ori-gin or course (such as ALCAPA) or in association with otherheart anomalies (such as transposition of the great arteries).These patients are managed surgically, usually with coronaryreimplantation. The incidence of coronary artery stenosisrequiring PCI in infancy occurs only as a rare complicationafter these coronary artery surgeries [1–6].We present a successful case of coronary artery stentingin an infant with ALCAPA with a significant postoperativestenosis.2. Case ReportOur patient was born overseas, in São Tomé and Principe, asmall island nation off the western coast of Africa. Despiteearly symptoms of overt heart failure, she was only evacu-ated to our country at the age of 18 months, weighing8.9 kg. Upon arrival, we made the echocardiographic-baseddiagnosis of an anomalous left coronary artery (LCA) fromthe pulmonary artery (ALCAPA) with a dilated and poorlyfunctioning left ventricle (LV). The patient underwentuneventful left coronary artery implantation in the left-facing aortic sinus. Due to persistent postoperative hemor-rhage around the site of LCA reimplantation, biological gluewas used, with success. However, in the postoperatoryperiod, there was no improvement of the LV function andsize, and the patient remained in low cardiac output. TheCT scan showed a significant left main coronary stenosisand ruled out intracoronary thrombosis.On the 4th postoperative day, the patient underwent car-diac catheterization (Figure 1). It ruled out left coronaryartery ostial narrowing at the anastomosis site but showedHindawiCase Reports in CardiologyVolume 2022, Article ID 3815465, 3 pageshttps://doi.org/10.1155/2022/3815465a severe rosary-bead-like stenosis (minimum diameter1.1mm) that involved the whole left main coronary arteryand extended into the ostia of the left anterior descendingand circumflex arteries. The heart team discussed the caseon-site, and the patient was refused for surgical reinterven-tion, and therefore, a PCI was attempted. Informed consentwas obtained prior to the procedure.We were unable to selectively catheterize the leftcoronary artery with several guiding catheters, which provedtoo large for the patient size. Therefore, we used a 4Frdiagnostic Judkins left 2.5 catheter to catheterize the leftcoronary artery with a 0.014″ hydrophilic coronary guide-wire, a Runthrough NS Hypercoat (Terumo InterventionalSystems, Somerset, NJ, USA), which was placed distally ina diagonal artery. We exchanged over-the-wire the diagnos-tic catheter for a 5Fr Judkins left 3.5 guiding catheter, whichwas left in the left-sided sinus of Valsalva. The coronary ste-nosis was inframillimetric, and the normal anterior descend-ing coronary artery measured 3mm. We implanted a 3 × 12mm Xience Sierra everolimus drug-eluting coronary stentsystem (Abbott Vascular, Santa Clara, CA, USA). Theimmediate postinterventional coronarography showed anoptimal angiographic result, with adequate distal flow toboth the anterior descending and circumflex coronaryarteries.The patient was placed under dual antiplatelet therapywith aspirin (5mg/kg/day) and clopidogrel (0.2mg/kg/d).Despite a widely patent flow in the coronary stent, well seenin serial transthoracic echocardiograms, the left ventriclefunction and size did not recover. The patient required aleft-ventricular assist device due to persistent low cardiacoutput syndrome. She eventually died, seven days later, withsepsis.3. DiscussionPostoperative LCA occlusion and persistent LV dysfunctionafter ALCAPA surgery are not uncommon [10, 11]. In ourcase, we speculate that external compression (due to the bio-logical glue, blood clots, or other mechanisms) or coronaryartery kinking due to surgical proximal segment mobiliza-tion may have caused the LCA stenosis. Another contribut-ing factor could be the presence of intracoronary (so-called“steal”) collaterals that may exist in ALCAPA and result incompetitive flow to the normal coronary artery system [12].We are aware that coronary artery stenting in this situa-tion is only tolerable as a bailout technique in a critically illinfant, especially considering the risks of stent thrombosisand dual antiplatelet antiaggregation in a small infant andthe fixed stent diameter which will not accompany thepatient’s somatic growth.There are many challenges associated with coronaryartery stenting in infants which make this a high-riskprocedure. All seven published case reports were technicallysuccessful but carried significant acute and short-term com-plications, including myocardial ischemia, need for extracor-poreal membrane oxygenator support, early in-stentthrombosis, and premature death. Several factors contrib-uted to these findings, including the adverse baseline clinicalsituation that leads to the cardiac catheterization, the smallpatient weight, the diminutive coronary diameter, and themultiple anatomic variants in a postoperative situation.Additionally, there is a limited availability of adequate coro-nary catheterization hardware, which is designed for adult-sized coronary arteries and vessels, that can be cumbersometo manage in small patients. The choice of bare metal stent(BMS) vs. drug-eluting stent (DES) is also up for debate,due to concerns about potential deleterious side effects ofsystemic circulation of antiproliferative drugs in smallpatients [13]. In the previous reports, only 2/7 stents wereDES, the remainder were BMS. Considering preliminarysafety data of neonatal DES in the ductus arteriosus, weopted for a recent generation of DES. There is also debateabout the optimal thromboprophylaxis regimen, which wasdual-antiplatelet therapy in 5/7 and single-antiplatelet (aspi-rin) in the remainder. We chose a dual-antiplatelet regimen,which is the recommended thromboprophylaxis for DES.We had no hemorrhagic complications. Finally, there is achallenge due to the understandable lack of coronary arteryexpertise of the pediatric interventionalists. In our case, itwas crucial to perform a four-hand procedure with an expe-rienced adult congenital heart disease interventionalist.Our case highlights the importance of collaborative workbetween adult and pediatric cardiologists that allows a(a) (b)Figure 1: On the left-hand-sided panel (a), the angiography reveals a significant stenosis of the left main coronary artery. The right-hand-sided panel (b) depicts an excellent angiographic result after stent implantation in the left main coronary artery, which is now widely patent.2 Case Reports in Cardiologypatient-focused treatment and some technical challenges ofthis challenging interventional technique in infants.Conflicts of InterestThe authors declare that there is no conflict of interest.Supplementary MaterialsSupplementaryMaterial (video legend): available at: https://drive.google.com/drive/folders/10W_b_ZjXjJS7ui5uhers6xJrQiGC_uc3?usp=sharing. Part 1: preintervention aortic root angiogra-phy, showing a right dominant system and a severe stenosis ofthe left main coronary artery. Part 2: 4Fr diagnostic Judkinsleft 2.5 catheter is used to catheterize the left coronary arterywith a 0.014″ hydrophilic coronary guidewire. Part 3: prein-tervention selective left coronary artery angiography, showinga severe rosary-bead-like stenosis that involved the whole leftmain coronary artery and extended into the ostia of the leftanterior descending and circumflex arteries. Parts 4 and 5:postintervention selective angiographies, showing an ade-quately placed stent in the left main coronary artery, with agood angiographic result. (Supplementary Materials)References[1] A. Bratincsak, A. Salkini, H. G. El-Said, and J. W. Moore, “Per-cutaneous stent implantation into coronary arteries ininfants,” Catheterization and Cardiovascular Interventions,vol. 79, no. 2, pp. 303–311, 2012.[2] A. Hallbergson and J. J. Rome, “Percutaneous left main coro-nary artery stent for acute myocardial ischemia after repairedALCAPA,” Catheterization and Cardiovascular Interventions,vol. 85, no. 6, pp. 1017–1020, 2015.[3] M. Schneider, W. Wiebe, V. Hraska, and P. Zartner, “Coro-nary interventions in congenital heart diseases: from pretermto young adult patients,” Journal of Interventional Cardiology,vol. 26, no. 3, pp. 287–294, 2013.[4] J. S. de Lezo, M. Pan, and C. Herrera, “Combined percutane-ous revascularization and cell therapy after failed repair ofanomalous origin of left coronary artery from pulmonaryartery,” Catheterization and Cardiovascular Interventions,vol. 73, no. 6, pp. 833–837, 2009.[5] G. S. Chrysant, D. Balzer, and M. Taniuchi, “Left main stemcoronary artery stenting in a 3-month-old child after anoma-lous left coronary artery from pulmonary artery repair,” Pedi-atric Cardiology, vol. 26, no. 5, pp. 734–736, 2005.[6] A. E. Schneider, J. N. Johnson, N. W. Taggart et al., “Percuta-neous coronary intervention in pediatric and adolescentpatients,” Congenital Heart Disease, vol. 9, no. 3, pp. 228–234, 2014.[7] A. A. Divekar, J. J. Lee, W. J. Tymchak, and J. M. Rutledge,“Percutaneous coronary intervention for extrinsic coronarycompression after pulmonary valve replacement,” Catheteri-zation and Cardiovascular Interventions, vol. 67, no. 3,pp. 482–484, 2006.[8] H. Muta and M. Ishii, “Percutaneous coronary interventionversus coronary artery bypass grafting for stenotic lesions afterKawasaki disease,” The Journal of Pediatrics, vol. 157, no. 1,pp. 120–126, 2010.[9] R. E. Shaddy, J. A. Revenaugh, G. S. Orsmond, and L. Y. Tani,“Coronary interventional procedures in pediatric heart trans-plant recipients with cardiac allograft vasculopathy,” TheAmerican Journal of Cardiology, vol. 85, no. 11, pp. 1370–1372, 2000.[10] A. Secinaro, H. Ntsinjana, O. Tann et al., “Cardiovascularmagnetic resonance findings in repaired anomalous left coro-nary artery to pulmonary artery connection (ALCAPA),” Jour-nal of Cardiovascular Magnetic Resonance, vol. 13, no. 1, p. 27,2011.[11] B. Castaldi, V. Vida, E. Reffo et al., “Speckle tracking inALCAPA patients after surgical repair as predictor of residualcoronary disease,” Pediatric Cardiology, vol. 38, no. 4, pp. 794–800, 2017.[12] M. A. Frommelt, E. Miller, J. Williamson, and S. Bergstrom,“Detection of septal coronary collaterals by color flow Dopplermapping is a marker for anomalous origin of a coronary arteryfrom the pulmonary artery,” Journal of the American Society ofEchocardiography, vol. 15, no. 3, pp. 259–263, 2002.[13] V. Aggarwal, G. S. Dhillon, D. J. Penny, S. T. Gowda, and A.M.Qureshi, “Drug-eluting stents compared with bare metal stentsfor stenting the ductus arteriosus in infants with ductal-dependent pulmonary blood flow,” The American Journal ofCardiology, vol. 124, no. 6, pp. 952–959, 2019.3Case Reports in Cardiology

A Rare but Real Necessity: Case Report of Coronary Artery Stenting in an Infant

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A Rare but Real Necessity: Case Report of Coronary Artery Stenting in an Infant

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