{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,4,23]],"date-time":"2025-04-23T00:40:08Z","timestamp":1745368808219,"version":"3.40.4"},"reference-count":12,"publisher":"Texas Heart Institute Journal","issue":"6","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2022,11,1]]},"abstract":"<jats:p>Anomalous origin of the left coronary artery from the opposite sinus of Valsalva with an intramural aortic course (L-ACAOS-IM) can cause syncope, sometimes as a prodrome of lethal events, including sudden cardiac death, in young athletes. The detailed mechanism of syncope in patients with L-ACAOS-IM is still unclear. This case report describes a 17-year-old boy who presented to the hospital because of syncope following chest pain with increasing frequency during exercise, such as playing soccer and running. In a treadmill exercise test, a decrease in blood pressure was seen (from 99\/56 mm Hg to 68\/38 mm Hg); chest pain and faintness accompanied by ST-segment elevation in lead aVR and ST-segment depression at other leads on electrocardiography were noted. These findings and symptoms disappeared spontaneously within a few minutes while clinicians prepared for emergency medications. Coronary computed tomography angiography (CCTA) showed that the origin of the left coronary artery (LCA) was the opposite sinus of Valsalva, and the course of the LCA was through the aortic wall toward the left coronary sinus. He was diagnosed with L-ACAOS-IM. After surgical treatment by unroofing the intramural part of the LCA and reconstructing a neo-ostium, he no longer experienced syncope during exercise. This case suggests that low cardiac output caused by myocardial ischemia, not life-threatening arrythmia, is a main mechanism of syncope in patients with L-ACAOS-IM. Consideration should be given to performing CCTA before an exercise stress test for young patients with syncope and chest pain to avoid the risk of severe myocardial ischemia.<\/jats:p>","DOI":"10.14503\/thij-21-7677","type":"journal-article","created":{"date-parts":[[2022,12,20]],"date-time":"2022-12-20T18:04:22Z","timestamp":1671559462000},"source":"Crossref","is-referenced-by-count":0,"title":["Repeated Syncope During Exercise as a Result of Anomalous Origin of Left Coronary Artery With Intramural Aortic Course in a Teenage Boy"],"prefix":"10.14503","volume":"49","author":[{"given":"Naofumi","family":"Amioka","sequence":"first","affiliation":[{"name":"Department of Cardiovascular Medicine, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kazufumi","family":"Nakamura","sequence":"additional","affiliation":[{"name":"Department of Cardiovascular Medicine, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Naoaki","family":"Matsuo","sequence":"additional","affiliation":[{"name":"Department of Cardiovascular Medicine, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Atsuyuki","family":"Watanabe","sequence":"additional","affiliation":[{"name":"Department of Cardiovascular Medicine, National Hospital Organization Okayama Medical Center, Okayama, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yasuhiro","family":"Kotani","sequence":"additional","affiliation":[{"name":"Department of Cardiovascular Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama, Japan"},{"name":"Department of Cardiovascular Surgery, Okayama University Hospital, Okayama, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shingo","family":"Kasahara","sequence":"additional","affiliation":[{"name":"Department of Cardiovascular Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama, Japan"},{"name":"Department of Cardiovascular Surgery, Okayama University Hospital, Okayama, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hiroshi","family":"Ito","sequence":"additional","affiliation":[{"name":"Department of Cardiovascular Medicine, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"5628","reference":[{"issue":"1","key":"i1526-6702-49-6-e217677-b1","series-title":"J Am Coll Cardiol.","first-page":"148","article-title":"Prospective echocardiographic diagnosis and surgical repair of anomalous origin of a coronary artery from the opposite sinus with an interarterial course","volume":"42","year":"2003","unstructured":"Frommelt\nPC,\n\n\nFrommelt\nMA,\n\n\nTweddell\nJS,\n\n\nJaquiss\nRDB.\n\n\nProspective echocardiographic diagnosis and surgical repair of anomalous origin of a coronary artery from the opposite sinus with an interarterial course.\nJ Am Coll Cardiol.2003;42(1):148\u2013154.\ndoi:10.1016\/s0735-1097(03)00503-5"},{"issue":"10","key":"i1526-6702-49-6-e217677-b2","series-title":"Circulation.","first-page":"1296","article-title":"Coronary artery anomalies: an entity in search of an identity","volume":"115","year":"2007","unstructured":"Angelini\nP.\n\n\nCoronary artery anomalies: an entity in search of an identity.\nCirculation.2007;115(10):1296\u20131305.\ndoi:10.1161\/CIRCULATIONAHA.106.618082"},{"issue":"11","key":"i1526-6702-49-6-e217677-b3","series-title":"Am J Med.","first-page":"1170","article-title":"Demographics and epidemiology of sudden deaths in young competitive athletes: from the United States National Registry","volume":"129","year":"2016","unstructured":"Maron\nBJ,\n\n\nHaas\nTS,\n\n\nAhluwalia\nA,\n\n\nMurphy\nCJ,\n\n\nGarberich\nRF.\n\n\nDemographics and epidemiology of sudden deaths in young competitive athletes: from the United States National Registry.\nAm J Med.2016;129(11):1170\u20131177.\ndoi:10.1016\/j.amjmed.2016.02.031"},{"issue":"3","key":"i1526-6702-49-6-e217677-b4","series-title":"Clin Auton Res.","first-page":"127","article-title":"Influence of age and gender on the occurrence and presentation of reflex syncope","volume":"18","year":"2008","unstructured":"Romme\nJJ,\n\n\nvan Dijk\nN,\n\n\nBoer\nKR,\n\n\n.\nInfluence of age and gender on the occurrence and presentation of reflex syncope.\nClin Auton Res.2008;18(3):127\u2013133.\ndoi:10.1007\/s10286-008-0465-0"},{"issue":"7","key":"i1526-6702-49-6-e217677-b5","series-title":"Hum Pathol.","first-page":"689","article-title":"Anomalous origin of coronary arteries and risk of sudden death: a study based on an autopsy population of congenital heart disease","volume":"29","year":"1998","unstructured":"Frescura\nC,\n\n\nBasso\nC,\n\n\nThiene\nG,\n\n\n.\nAnomalous origin of coronary arteries and risk of sudden death: a study based on an autopsy population of congenital heart disease.\nHum Pathol.1998;29(7):689\u2013695.\ndoi:10.1016\/s0046-8177(98)90277-5"},{"issue":"3","key":"i1526-6702-49-6-e217677-b6","series-title":"J Am Coll Cardiol.","first-page":"640","article-title":"Sudden cardiac death associated with isolated congenital coronary artery anomalies","volume":"20","year":"1992","unstructured":"Taylor\nAJ,\n\n\nRogan\nKM,\n\n\nVirmani\nR.\n\n\nSudden cardiac death associated with isolated congenital coronary artery anomalies.\nJ Am Coll Cardiol.1992;20(3):640\u2013647.\ndoi:10.1016\/0735-1097(92)90019-j"},{"issue":"4","key":"i1526-6702-49-6-e217677-b7","series-title":"Am Heart J.","first-page":"428","article-title":"Anomalous right or left coronary artery from the contralateral coronary sinus: \u201chigh-risk\u201d abnormalities in the initial coronary artery course and heterogeneous clinical outcomes","volume":"133","year":"1997","unstructured":"Taylor\nAJ,\n\n\nByers\nJP,\n\n\nCheitlin\nMD,\n\n\nVirmani\nR.\n\n\nAnomalous right or left coronary artery from the contralateral coronary sinus: \u201chigh-risk\u201d abnormalities in the initial coronary artery course and heterogeneous clinical outcomes.\nAm Heart J.1997;133(4):428\u2013435.\ndoi:10.1016\/s0002-8703(97)70184-4"},{"issue":"2","key":"i1526-6702-49-6-e217677-b8","series-title":"Eur Heart J Cardiovasc Imaging.","first-page":"224","article-title":"Anomalous origin of the coronary artery arising from the opposite sinus: prevalence and outcomes in patients undergoing coronary CTA","volume":"18","year":"2017","unstructured":"Cheezum\nMK,\n\n\nGhoshhajra\nB,\n\n\nBittencourt\nMS,\n\n\n.\nAnomalous origin of the coronary artery arising from the opposite sinus: prevalence and outcomes in patients undergoing coronary CTA.\nEur Heart J Cardiovasc Imaging.2017;18(2):224\u2013235.\ndoi:10.1093\/ehjci\/jev323"},{"issue":"6","key":"i1526-6702-49-6-e217677-b9","series-title":"J Thorac Cardiovasc Surg.","first-page":"1440","article-title":"Expert consensus guidelines: anomalous aortic origin of a coronary artery","volume":"153","year":"2017","unstructured":"Brothers\nJA,\n\n\nFrommelt\nMA,\n\n\nJaquiss\nRDB,\n\n\nMyerburg\nRJ,\n\n\nFraser\nCD\nJr,\n\n\nTweddell\nJS.\n\n\nExpert consensus guidelines: anomalous aortic origin of a coronary artery.\nJ Thorac Cardiovasc Surg.2017;153(6):1440\u20131457.\ndoi:10.1016\/j.jtcvs.2016.06.066"},{"issue":"7","key":"i1526-6702-49-6-e217677-b10","series-title":"Clin Case Rep.","first-page":"1371","article-title":"Anomalous aortic origin of the right coronary artery with functional ischemia determined with fractional flow reserve derived from computed tomography","volume":"6","year":"2018","unstructured":"Miki\nT,\n\n\nMiyoshi\nT,\n\n\nWatanabe\nA,\n\n\nOsawa\nK,\n\n\nAmioka\nN,\n\n\nIto\nH.\n\n\nAnomalous aortic origin of the right coronary artery with functional ischemia determined with fractional flow reserve derived from computed tomography.\nClin Case Rep.2018;6(7):1371\u20131372.\ndoi:10.1002\/ccr3.1582"},{"issue":"2","key":"i1526-6702-49-6-e217677-b11","series-title":"Catheter Cardiovasc Interv.","first-page":"313","article-title":"Anatomic spectrum of left coronary artery anomalies and associated mechanisms of coronary insufficiency","volume":"92","year":"2018","unstructured":"Angelini\nP,\n\n\nUribe\nC.\n\n\nAnatomic spectrum of left coronary artery anomalies and associated mechanisms of coronary insufficiency.\nCatheter Cardiovasc Interv.2018;92(2):313\u2013321.\ndoi:10.1002\/ccd.27656"},{"issue":"2","key":"i1526-6702-49-6-e217677-b12","series-title":"Tex Heart Inst J.","first-page":"168","article-title":"In syncope or sudden death from coronary artery anomalies, hypotension and bradycardia are more frequent than ventricular fibrillation","volume":"47","year":"2020","unstructured":"Angelini\nP.\n\n\nIn syncope or sudden death from coronary artery anomalies, hypotension and bradycardia are more frequent than ventricular fibrillation.\nTex Heart Inst J.2020;47(2):168\u2013169.\ndoi:10.14503\/THIJ-19-7105"}],"container-title":["Texas Heart Institute Journal"],"original-title":[],"link":[{"URL":"https:\/\/thij.kglmeridian.com\/view\/journals\/thij\/49\/6\/article-e217677.xml","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/meridian.allenpress.com\/thij\/article-pdf\/doi\/10.14503\/THIJ-21-7677\/3415211\/i1526-6702-49-6-e217677.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/thij.kglmeridian.com\/downloadpdf\/journals\/thij\/49\/6\/article-e217677.xml","content-type":"text\/html","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/thij.kglmeridian.com\/downloadpdf\/journals\/thij\/49\/6\/article-e217677.xml","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,4,23]],"date-time":"2025-04-23T00:02:30Z","timestamp":1745366550000},"score":1,"resource":{"primary":{"URL":"https:\/\/thij.kglmeridian.com\/view\/journals\/thij\/49\/6\/article-e217677.xml"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,11,1]]},"references-count":12,"journal-issue":{"issue":"6"},"URL":"https:\/\/doi.org\/10.14503\/thij-21-7677","relation":{},"ISSN":["1526-6702","0730-2347"],"issn-type":[{"type":"electronic","value":"1526-6702"},{"type":"print","value":"0730-2347"}],"subject":[],"published":{"date-parts":[[2022,11,1]]}}}