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Reimplantation of anomalous right coronary artery arising from the pulmonary trunk into the aorta is low risk and good midterm result

Ivanov AS

Federal Research Centre of Transplantology and Artificial Organs named after V.I. Shumakov, Moscow, Russian Federation.

E-mail : bhuvaneswari.bibleraaj@uhsm.nhs.uk

Gureev SV

Federal Research Centre of Transplantology and Artificial Organs named after V.I. Shumakov, Moscow, Russian Federation.

Glamazda SV

Federal Research Centre of Transplantology and Artificial Organs named after V.I. Shumakov, Moscow, Russian Federation.

Ostroumov EN

Federal Research Centre of Transplantology and Artificial Organs named after V.I. Shumakov, Moscow, Russian Federation.

Lugovskiy MK

Federal Research Centre of Transplantology and Artificial Organs named after V.I. Shumakov, Moscow, Russian Federation.

DOI: 10.15761/CDM.1000144

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Abstract

This report describes results  of right coronary artery reimplantation with tomography, myocardial perfusion imaging and diagnostic coronary angiography evaluation before and 1 year after operation on a 9 years old boy with  anomalous right coronary artery  from pulmonary artery.​

Key words

Anomalous right coronary artery; reimplantation.

Introduction

The clinical profile and the natural history of Anomalous right coronary artery (RCA) from pulmonary artery (ARCAPA) are not well delineated, and the information is derived mostly from individual case reports, [2,3,4]. This report describes results  of  right coronary artery (RCA) reimplantation with tomography, myocardial perfusion imaging and diagnostic coronary angiography evaluation before and 1 year after operation on a 9 years old boy with  ARCAPA.

Case Report

The 9 years old boy,  was hospitalized for evaluation of a heart murmur  and diagnostics of coronary-pulmonary fistula.  His only complaint was  fatigue during the physical exercises.

Spiral contrast computed tomography: Left coronary artery (LCA) located typically. LCA trunk diameter-7 mm. Anterior descending artery diameter-6 mm. Left circumflex artery diameter-5 mm, both located typically. RCA originates from the trunk of pulmonary artery (Figure 1B). Diagnostic coronary angiography confirmed these findings and demonstrated that RCA was dilated along all its length and the retrograde flow of contrasted blood from LCA filled RCA through the collateral system and went further into pulmonary artery (Figure 1А). Myocardial perfusion imaging with ECG-gated single-photon emission computed tomography (SPECT) with 170 mBk MIBI-Tc99m: left ventricular myocardium with the areas of mild hypoperfusion in the anterior septal zone (Figure 2C).

Figure 1.
A)
coronary angiography before operation, went into pulmonary artery indicated by arrow.

B) Computed tomography before operation. RCA – right coronary artery, LCA – left coronary artery, PA – pulmonary artery.

Figure 2.

A. Computed tomography 5 days after RCA re-implantation.

B. coronary angiography of RCA 1 year after RCA reimplantation; C/D - Myocardial perfusion imaging

C. (before operation) - left ventricular myocardium with the areas of mild hypoperfusion in the anterior septal zone.

D. (after operation) - Areas of LV anterior and septal walls hypoperfusion on myocardial perfusion imaging with ECG-gated single-photon emission computed tomography disappeared after RCA reimplantation.

Оperation

RCA trunk  was decided to fulfil translocation of the RCA mouth from the pulmonary artery to ascending aorta without extracorporeal circulation on the beating heart. RCA mouth  has been ligated, cut, and  subsequently RCA was re-implanted into aorta. Antegrade flow through it was documented.  The result of operation see on Figure 2А.

Result

Areas of Left Ventricular anterior and septal walls hypoperfusion on myocardial perfusion imaging  disappeared after RCA reimplantation (Figure 2 D).

One year after the operation: According to his mother,  he became more active than before the operation.  Diagnostic coronary angiography: contrasted blood flow through the arteries was normal. Right coronary artery originates from the aorta, its  proximal part  was dilated to 11 mm (from 7 mm), there was an area of  4 mm stenosis on the border of the medium and proximal parts of the artery. The large proximal branch RCA was obliterated. (Indicated by arrow) (Figure 2 B).

Discussion

ARCAPA is a very rare congenital  anomaly first described in 1885 by Brooks, [1]. This condition, when not combined with other congenital heart defects, is often asymptomatic and is found incidentally. However, there are some patients with ARCAPA, who suffer from exertion angina, and the cases of sudden death were reported as well [5]. When RCA originates from the pulmonary artery, steal syndrome may cause exercise-induced myocardial ischemia. This may be the mechanism of sudden death, and if it is true, then surgical correction appears warranted.

In our case this approach provided for the clinical improvement, normalization of myocardial perfusion and diastolic function of both ventriculars and reduction of LV asynchrony and increase in EF of RV from 20 to 34% in one year.  

In one year after RCA translocation the dilatation of its proximal part with its subsequent stenosis was detected. Similar situation was  described by  Veselka J, [4]. We assume that this dilatation was due to the redirection of the blood flow through RCA, and the pressure increase within RCA, which has been previously adapted to the low pressure. Vice versa, - the LCA diameter has decreased in comparison with preoperative data. The decsrease of LCA diameter was probably due to the improvement of capillary blood flow and the decrease in vascular resistance.

Conclusion

Although most of the patients with ARCAPA remain asymptomatic, reimplantation of the RCA into the aorta may be recommended to prevent myocardial hypoperfusion and its consequences.

References

  1. Brooks HSJ (1885) Two Cases of an Abnormal Coronary Artery of the Heart Arising from the Pulmonary Artery. J Anat Physiol 20(Pt 1): 26-29. [crossref]
  2. Williams IA, Gersony WM, Hellenbrand WE (2006) Anomalous right coronary artery arising from the pulmonary artery: a report of 7 cases and a review of the literature. Am Heart J 152:1004. [crossref]
  3. Refatllari A, Likaj E, Dumani S, Hasimi E, and Goda A (2015) Surgical Treatment of Anomalous Origin of Right Coronary Artery in a Patient with Mitral Stenosis. Open Access Maced J Med Sci 15;4(1):131-4. [crossref]
  4. Veselka J, Widimsky´ P, Kautzner J (2003) Reimplantation of anomalous right coronary artery arising from the pulmonary trunk leading to normal coronary flow reserve late after surgery. Ann Thorac Surg 76: 1287-1289. [crossref]
  5. Click RL, Holmes DR Jr, Vlietstra RE, Kosinski AS, Kronmal RA (1989) Anomalous coronary arteries: location, degree of atherosclerosis and effect on survival: a report from the Coronary Artery Surgery Study. J Am Coll Cardiol 13:531-537. [crossref]

Editorial Information

Editor-in-Chief

Richard Kones
Cardiometabolic Research Institute

Article Type

Case Report

Publication history

Received date: April 22, 2017
Accepted date: May 29, 2017
Published date: May 31, 2017

Copyright

© 2017 Ivanov AS. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

Citation

Ivanov AS, Gureev SV, Glamazda SV, Ostroumov EN, Lugovskiy MK (2017) Reimplantation of anomalous right coronary artery arising from the pulmonary trunk into the aorta is low risk and good midterm result . Cardiovasc Disord Med 2: DOI: 10.15761/CDM.1000144

Corresponding author

Sergey V Gureev

Federal reseach centre of transplantology named after Shumakov Moscow, Moscow Russian Federation

E-mail : bhuvaneswari.bibleraaj@uhsm.nhs.uk

Figure 1.
A)
coronary angiography before operation, went into pulmonary artery indicated by arrow.

B) Computed tomography before operation. RCA – right coronary artery, LCA – left coronary artery, PA – pulmonary artery.

Figure 2.

A. Computed tomography 5 days after RCA re-implantation.

B. coronary angiography of RCA 1 year after RCA reimplantation; C/D - Myocardial perfusion imaging

C. (before operation) - left ventricular myocardium with the areas of mild hypoperfusion in the anterior septal zone.

D. (after operation) - Areas of LV anterior and septal walls hypoperfusion on myocardial perfusion imaging with ECG-gated single-photon emission computed tomography disappeared after RCA reimplantation.