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
Both the anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) and unilateral absence of the pulmonary artery (UAPA) are rare congenital malformations, ALCAPA accompanied with UAPA is extremely rare. Here, we reported a middle-aged man admitted to our department for evaluat...
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| Format: | Article |
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Frontiers Media S.A.
2023-04-01
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| Series: | Frontiers in Cardiovascular Medicine |
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| Online Access: | https://www.frontiersin.org/articles/10.3389/fcvm.2023.1160893/full |
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| author | Dong Yi Juan Xia Xiang Zhou Chengwei Liu Li Liu Hua Yan Xiaojing Ma |
| author_facet | Dong Yi Juan Xia Xiang Zhou Chengwei Liu Li Liu Hua Yan Xiaojing Ma |
| author_sort | Dong Yi |
| collection | DOAJ |
| description | Both the anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) and unilateral absence of the pulmonary artery (UAPA) are rare congenital malformations, 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 did not 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 into the pulmonary artery and dilated right coronary artery (RCA), which supported but did not 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 (LMCA) arising from the pulmonary artery, and it incidentally unveiled another rare congenital malformation of UAPA. The patient underwent surgical correction of ALCAPA by reimplantation of the 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 non-invasive 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 an adult patient. |
| first_indexed | 2024-04-09T17:11:32Z |
| format | Article |
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| language | English |
| last_indexed | 2024-04-09T17:11:32Z |
| publishDate | 2023-04-01 |
| publisher | Frontiers Media S.A. |
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| series | Frontiers in Cardiovascular Medicine |
| spelling | doaj.art-7457701da0024fc0b7b821785d326bfb2023-04-20T05:53:25ZengFrontiers Media S.A.Frontiers in Cardiovascular Medicine2297-055X2023-04-011010.3389/fcvm.2023.11608931160893Case 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 patientDong Yi0Juan Xia1Xiang Zhou2Chengwei Liu3Li Liu4Hua Yan5Xiaojing Ma6Department of Cardiology, Wuhan Asia Heart Hospital, Wuhan, ChinaDepartment of Echocardiography, Wuhan Asia Heart Hospital, Wuhan, ChinaDepartment of Cardiac Surgery, Wuhan Asia Heart Hospital, Wuhan, ChinaDepartment of Cardiology, Wuhan Asia Heart Hospital, Wuhan, ChinaDepartment of Cardiology, Wuhan Asia Heart Hospital, Wuhan, ChinaDepartment of Cardiology, Wuhan Asia Heart Hospital, Wuhan, ChinaDepartment of Echocardiography, Wuhan Asia Heart Hospital, Wuhan, ChinaBoth the anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) and unilateral absence of the pulmonary artery (UAPA) are rare congenital malformations, 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 did not 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 into the pulmonary artery and dilated right coronary artery (RCA), which supported but did not 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 (LMCA) arising from the pulmonary artery, and it incidentally unveiled another rare congenital malformation of UAPA. The patient underwent surgical correction of ALCAPA by reimplantation of the 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 non-invasive 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 an adult patient.https://www.frontiersin.org/articles/10.3389/fcvm.2023.1160893/fullanomalous originleft coronary arterypulmonary arteryechocardiographycoronary angiographymultidetector computed tomographic angiogram |
| spellingShingle | Dong Yi Juan Xia Xiang Zhou Chengwei Liu Li Liu Hua Yan Xiaojing Ma 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 Frontiers in Cardiovascular Medicine anomalous origin left coronary artery pulmonary artery echocardiography coronary angiography multidetector computed tomographic angiogram |
| 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 |
| title_full | 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 |
| title_fullStr | 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 |
| title_full_unstemmed | 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 |
| title_short | 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 |
| title_sort | 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 |
| topic | anomalous origin left coronary artery pulmonary artery echocardiography coronary angiography multidetector computed tomographic angiogram |
| url | https://www.frontiersin.org/articles/10.3389/fcvm.2023.1160893/full |
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