AUTHOR=Yi Dong , Xia Juan , Zhou Xiang , Liu Chengwei , Liu Li , Yan Hua , Ma Xiaojing 

TITLE=Case report: A rare case of anomalous origin of the left coronary artery from the pulmonary artery accompanied with unilateral absence of pulmonary artery in an adult patient

JOURNAL=Frontiers in Cardiovascular Medicine

VOLUME=Volume 10 - 2023

YEAR=2023

URL=https://www.frontiersin.org/journals/cardiovascular-medicine/articles/10.3389/fcvm.2023.1160893

DOI=10.3389/fcvm.2023.1160893

ISSN=2297-055X

ABSTRACT=Both anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) and unilateral absence of the pulmonary artery (UAPA) are rare congenital malformation, ALCAPA accompanied with UAPA is extremely rare. Here we reported a middle-aged man admitted to our department for evaluation of chest pain during exercise. Physical examination and lab tests didn’t unveil obvious abnormality, however, transthoracic echocardiogram (TTE) revealed multivessel myocardial collateral blood flow signals in the left ventricular wall and ventricular septum, a shunting flow from the left coronary artery (LCA) into the pulmonary artery (PA) and dilated right coronary artery (RCA), which supported but didn’t confirm the diagnosis of ALCAPA. Coronary angiography (CAG) showed an absent left coronary ostium and a dilated RCA, with extensive collaterals supplying the left coronary system. Multidetector computed tomography angiography (MDCTA) was then performed and revealed the anomalous origin of the left main coronary artery arising from the pulmonary artery, and incidentally unveiled another rare congenital malformation of unilateral absence of the pulmonary artery (UAPA). The patient underwent surgical correction of ALCAPA by re-implantation of the left main coronary artery (LMCA) to the aorta, without surgical treatment of UAPA. The patient had been in good clinical condition and remained angina-free with good exercise tolerance during follow-up (~ 6 months so far). In this case, we discussed the diagnostic value of TTE, CAG and MDCTA on rare abnormalities as ALCAPA and UAPA. We highlighted the role of multiple noninvasive imaging modalities in diagnosing rare causes of angina in adult patients, and the importance of careful examination in avoiding misdiagnosis. To our best knowledge, this is the first report of ALCAPA accompanied with UAPA in adult patient.