Abstract
Anomalous origin of a coronary artery from the pulmonary artery (PA) is a rare congenital anomaly with the most common pattern being an anomalous left coronary artery (LCA) from the pulmonary artery (ALCAPA). A very rare pattern is the anomalous origin of a single coronary artery from the pulmonary artery (ASCAPA) wherein the single coronary ostium supplies both the right and left coronary systems with profound myocardial ischemia developing once PA pressures begin to fall after birth. Previous reports of this anomaly have all been based on post-mortem findings or pre-mortem angiographic diagnosis [1,2,3]. Notably, these children often present in extremis due to cardiogenic shock. Institution of veno-arterial extracorporeal life support can be catastrophic as it would lead to myocardial ischemia due to PA decompression. We present here the first echocardiographic-only diagnosis of ASCAPA in a 2-month-old child with the use of bubble contrast echocardiography to help confirm the diagnosis. The patient was resuscitated from a cardiac arrest on arrival with subsequent echocardiographic-only diagnosis. Surgical repair was undertaken with administration of cardioplegia into the pulmonary root with snaring of the branch PAs, and re-implantation of the single coronary to the aorta. At 3-month follow-up, he is thriving clinically with echocardiogram showing improving—though still somewhat depressed—left ventricular function.
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Nikhil Thatte declares that he has no conflict of interest. Manouk Kirakosian declares that he has no conflict of interest. Aditya Kaza declares that he has no conflict of interest. Kevin Friedman declares that he has no conflict of interest.
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Movie 1: High parasternal views demonstrate a coronary ostium arising from the rightward aspect of the main pulmonary artery and bifurcating into two vessels. There is antegrade flow from the pulmonary artery (PA) into both coronary vessels seen by color Doppler imaging which is contrary to what is expected in ALCAPA wherein there is retrograde flow in the left coronary draining into the PA. Supplementary file1 (AVI 63904 kb)
Movie 2: Agitated saline contrast imaging in a high parasternal view shows the presence of saline microbubbles in both the left coronary artery (LCA) and right coronary artery (RCA). No saline microbubbles were seen in the left heart (not depicted here). Supplementary file2 (MP4 14925 kb)
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Thatte, N., Kirakosian, M., Kaza, A. et al. Echocardiographic Diagnosis of Anomalous Single Coronary Artery from the Pulmonary Artery: Use of Bubble Contrast Echocardiography. Pediatr Cardiol 41, 215–216 (2020). https://doi.org/10.1007/s00246-019-02265-2
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DOI: https://doi.org/10.1007/s00246-019-02265-2