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Successful everolimus-eluting stent implantation into the left main trunk stenosis in the anomalous coronary artery after neo-aortic valve replacement in a 6-year-old boy

Part of: Interventional Cardiology

Published online by Cambridge University Press:  04 February 2019

Dai Asada ,
Hideshi Tomita  and
Takanari Fujii
Dai Asada*
Affiliation:
Department of Pediatric Cardiology, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kyoto, Japan
Hideshi Tomita
Affiliation:
Pediatric Heart Disease & Adult Congenital Heart Disease Center, Showa University Hospital, Shinagawa-ku, Tokyo, Japan
Takanari Fujii
Affiliation:
Pediatric Heart Disease & Adult Congenital Heart Disease Center, Showa University Hospital, Shinagawa-ku, Tokyo, Japan
*
Author for correspondence: D. Asada, 465 Kajiicho, Kawaramachi-Hirokoji, Kamigyo-ku, Kyoto 602-8566, Japan. Tel: +81 75 251 5832, Fax: +81 75 251 5833, E-mail: asadadai@koto.kpu-m.ac.jp
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Abstract

CHDs occasionally have coronary complications; however, stent implantation is technically difficult in small children. We reported a successful drug-eluting stent implantation into the congenital anomalous coronary artery in a 6-year-old boy. This treatment is useful for rescuing coronary stenosis, and dual antiplatelet therapy is important to prevent stent restenosis.

Keywords

Drug-eluting stentNorwood operationneo-aortic valve replacementanomalous coronary artery
Type
Brief Report
Information
Cardiology in the Young , Volume 29 , Issue 3 , March 2019 , pp. 448 - 450
Copyright
© Cambridge University Press 2019 

Despite several case reports describing successful deployment with acceptable short-term experiences in small children,Reference Kaichi, Doi, Tsurumi and Heike 1 coronary stent implantation is generally not considered a standard treatment option owing to its technical difficulties. It becomes more difficult in the anomalous coronary artery.

Here, we reported a successful stent deployment into the left main trunk stenosis in a 6-year-old boy with coronary artery angiectopia after neo-aortic valve replacement.

Case report

The patient was a 6-year-old boy weighing 20 kg. He was diagnosed with false Taussig–Bing anomaly, right ventricular hypoplasia, and aortic arch interruption and had Shaher type 9 coronary arterial anatomy (Fig 1a).Reference Shaher and Puddu 2 He underwent bilateral pulmonary artery banding, Norwood operation, and bidirectional cavo-pulmonary shunt in stages at his previous hospital. At the age of 1-year, he was referred to our hospital for total cavo-pulmonary connection; however, transthoracic echocardiography revealed severe neo-aortic regurgitation. He underwent total cavo-pulmonary connection at the age of 2 years, but the neo-aortic regurgitation gradually worsened and advanced atrioventricular block emerged. Neo-aortic valve replacement using 21-mm-diameter On-X valve (CryoLife Inc., Kennesaw, GA) and pacemaker implantation were performed at the age of 6 years. After this surgery, transthoracic echocardiography indicated low ejection fraction of 25% by Simpson method and hypokinesis of the anterior and lateral left ventricular wall. Electrocardiography showed ST-T alteration and Q wave in the precordial leads (Fig 2a), indicating ischemic change. We performed coronary angiography to investigate the ischemia. The standard view was unable to clearly detect the coronary orifice, but the coronary anatomy was well detected in the caudal view (Fig 1b). Coronary angiography revealed 90% stenosis of the left main trunk compressed by the mechanical valve; this stenosis was considered the cause of cardiac dysfunction. We decided to implant a drug-eluting stent to rescue the coronary blood flow. Informed consent was obtained from the parents.

Figure 1 The coronary artery in this case. (a) Schematic view of Shaher type 9; the LMT runs along the back of the pulmonary valve. (b) Coronary angiography revealed 90% stenosis of the LMT. The white arrow indicates stenosis. AV=aortic valve, LAD=left anterior descending, LCX=left circumflex, LMT=left main trunk, PV=pulmonary valve, RCA=right coronary artery.

Figure 2 ECG and coronary angiogram after AVR. (a) ECG showed ST-T alteration and Q wave in the precordial leads. (b) Stent was sufficiently dilated in good position. The white arrow indicated the restoration of blood flow. (c) Angiogram at 4 months after stent deployment revealed complete stent patency. AVR=aortic valve replacement, ECG=electrocardiogram, LAD=left anterior descending, LCX=left circumflex, LMT=left main trunk.

Stent implantation was performed under general anaesthesia. A right femoral vascular access was established, and 100 U/kg of intra-arterial heparin was administered. A 6-F Judkins left guiding catheter (Heartrail II; Terumo Corp, Tokyo) was advanced into the orifice of the left coronary artery, and a 0.014 in. guidewire (SION blue; Asahi Intecc Co, Nagoya) was inserted into the left main trunk. Intravascular ultrasonography (OptiCross™; Boston Scientific Corp., Marlborough, MA, United States of America) indicated a minimal lumen diameter of 0.4 mm and reference vessel diameter of 2.5 mm. Angiography revealed a stenosis length of 14 mm. We chose a 2.5–18 mm drug-eluting stent (XIENCE ALPINE; Abbott Vascular, Chicago, IL, United States of America) and deployed it at 10 atm. Post-dilation was performed using a 2.5–10 mm balloon catheter (Raiden 3; KANEKA MEDIX, Osaka) at 24 atm. Final angiography showed adequate dilation and good stent position (Fig 2b). No ischemic events occurred during the procedure.

After this procedure, dual antiplatelet therapy with aspirin and cilostazol was started. Follow-up angiography was performed at 4 months after stent implantation. All angiograms revealed complete stent patency (Fig 2c) and the ejection fraction by Simpson method using transthoracic echocardiography improved to 34%.

Discussion

We could successfully perform coronary stent deployment into the anomalous coronary artery using the caudal view in complex CHDs.

Norwood operation is usually performed to make a new aortic root using the pulmonary artery for the hypoplastic aorta. In this operation, the pulmonary valve is used as the neo-aortic valve. After this operation, neo-aortic regurgitation is a concern because exposure to systemic blood pressure or annular distortion causes functional deterioration in the pulmonary valve. Mechanical valve replacement is occasionally required,Reference Shimada, Hoashi, Kagisaki, Shiraishi, Yagihara and Ichikawa 3 but carries the risk of coronary artery compression because of abnormal anatomic spatial arrangement.Reference Hamzeh, El-Said and Moore 4 We considered reimplantation of smaller mechanical valve; however, previous reports have recommended an average diameter of 23 mm in children and showed the risk of aortic valve replacement under low cardiac function with coronary stenosis.Reference Arnold, Ley-Zaporozhan and Ley 5 , Reference Powell, Tunick and Rosenzweig 6 We performed coronary stent implantation for the left main trunk stenosis instead of reimplantation of smaller mechanical valve.

Stent deployment in children mainly poses the risk of restenosis and acute thrombotic stent occlusion. However, coronary stents have been innovated from bare metal stents to drug-eluting stents, improving the risk of stent thrombosis.Reference Palmerini, Biondi-Zoccai and Della Riva 7 Furthermore, Müller et al showed that the serum everolimus level after everolimus-eluting stent deployment is far below the immunosuppressive therapeutic range in infants.Reference Müller, Krause, Paul and Schneider 8 The results of these past reports persuaded us to select everolimus-eluting stent.

Dual antiplatelet therapy using aspirin and clopidogrel or cilostazol after drug-eluting stent deployment is not only a well-recognized supportive care in adults but also effective in reducing the probability of thrombotic events and the restenosis rate in small children.Reference Ueda, Kido and Matsuhisa 9 This patient had been taking warfarin and aspirin to prevent mechanical valve thrombosis. We added cilostazol to prevent restenosis, and follow-up angiography showed complete stent patency. In addition, Bratincsák et al reported that maintaining dual antiplatelet therapy for at least 12 months is crucial in infants after coronary stenting unless contraindications exist.Reference Bratincsák, Salkini, El-Said and Moore 10 We plan to continue this treatment for at least 12 months.

These findings suggest that everolimus-eluting stent implantation is useful in rescuing coronary stenosis in small children with anomalous coronary artery.

Acknowledgements

We thank Dr Naoei Isomura for her help with the coronary intervention and helpful insights.

Financial support

This research received no specific grant from any funding agency, or from commercial or not-for-profit sectors.

Conflicts of interest

None.

Footnotes

Cite this article: Asada D, Tomita H, Fujii T. (2019) Successful everolimus-eluting stent implantation into the left main trunk stenosis in the anomalous coronary artery after neo-aortic valve replacement in a 6-year-old boy. Cardiology in the Young29: 448–450. doi: 10.1017/S1047951118002512

References

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Figure 0

Figure 1 The coronary artery in this case. (a) Schematic view of Shaher type 9; the LMT runs along the back of the pulmonary valve. (b) Coronary angiography revealed 90% stenosis of the LMT. The white arrow indicates stenosis. AV=aortic valve, LAD=left anterior descending, LCX=left circumflex, LMT=left main trunk, PV=pulmonary valve, RCA=right coronary artery.

Figure 1

Figure 2 ECG and coronary angiogram after AVR. (a) ECG showed ST-T alteration and Q wave in the precordial leads. (b) Stent was sufficiently dilated in good position. The white arrow indicated the restoration of blood flow. (c) Angiogram at 4 months after stent deployment revealed complete stent patency. AVR=aortic valve replacement, ECG=electrocardiogram, LAD=left anterior descending, LCX=left circumflex, LMT=left main trunk.