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<article article-type="case-report" xmlns:mml="http://www.w3.org/1998/Math/MathML" xmlns:xlink="http://www.w3.org/1999/xlink" xml:lang="EN">
<front>
<journal-meta>
<journal-id journal-id-type="publisher-id">Front. Cardiovasc. Med.</journal-id>
<journal-title>Frontiers in Cardiovascular Medicine</journal-title>
<abbrev-journal-title abbrev-type="pubmed">Front. Cardiovasc. Med.</abbrev-journal-title>
<issn pub-type="epub">2297-055X</issn>
<publisher>
<publisher-name>Frontiers Media S.A.</publisher-name>
</publisher>
</journal-meta>
<article-meta>
<article-id pub-id-type="doi">10.3389/fcvm.2024.1504835</article-id>
<article-categories>
<subj-group subj-group-type="heading">
<subject>Cardiovascular Medicine</subject>
<subj-group>
<subject>Case Report</subject>
</subj-group>
</subj-group>
</article-categories>
<title-group>
<article-title>Uncommon presentation of left main congenital coronary aneurysm: a rare case report</article-title>
</title-group>
<contrib-group>
<contrib contrib-type="author"><name><surname>Xu</surname><given-names>Peng</given-names></name>
<xref ref-type="aff" rid="aff1"><sup>1</sup></xref>
<role content-type="https://credit.niso.org/contributor-roles/conceptualization/"/><role content-type="https://credit.niso.org/contributor-roles/writing-original-draft/"/><role content-type="https://credit.niso.org/contributor-roles/writing-review-editing/"/></contrib>
<contrib contrib-type="author"><name><surname>Zhong</surname><given-names>Shili</given-names></name>
<xref ref-type="aff" rid="aff2"><sup>2</sup></xref><uri xlink:href="https://loop.frontiersin.org/people/2857714/overview"/><role content-type="https://credit.niso.org/contributor-roles/data-curation/"/><role content-type="https://credit.niso.org/contributor-roles/visualization/"/><role content-type="https://credit.niso.org/contributor-roles/writing-review-editing/"/></contrib>
<contrib contrib-type="author" corresp="yes"><name><surname>Tan</surname><given-names>Hui</given-names></name>
<xref ref-type="aff" rid="aff3"><sup>3</sup></xref>
<xref ref-type="corresp" rid="cor1">&#x002A;</xref>
<role content-type="https://credit.niso.org/contributor-roles/writing-original-draft/"/><role content-type="https://credit.niso.org/contributor-roles/writing-review-editing/"/></contrib>
</contrib-group>
<aff id="aff1"><label><sup>1</sup></label><institution>Rehabilitation Medicine Department, Army Medical Center of PLA</institution>, <addr-line>Chongqing</addr-line>, <country>China</country></aff>
<aff id="aff2"><label><sup>2</sup></label><institution>Department of Intensive Care Medicine, Army Medical Center of PLA</institution>, <addr-line>Chongqing</addr-line>, <country>China</country></aff>
<aff id="aff3"><label><sup>3</sup></label><institution>Health Management Center, The Third Affiliated Hospital of Chongqing Medical University</institution>, <addr-line>Chongqing</addr-line>, <country>China</country></aff>
<author-notes>
<fn fn-type="edited-by"><p><bold>Edited by:</bold> Tommaso Gori, Johannes Gutenberg University Mainz, Germany</p></fn>
<fn fn-type="edited-by"><p><bold>Reviewed by:</bold> Ivan Ili&#x0107;, Institute for Cardiovascular Diseases Dedinje, Serbia</p>
<p>Denyan Mansuroglu, Istanbul Yeni Yuzyil University, T&#x00FC;rkiye</p></fn>
<corresp id="cor1"><label>&#x002A;</label><bold>Correspondence:</bold> Hui Tan <email>328602800@qq.com</email></corresp>
</author-notes>
<pub-date pub-type="epub"><day>02</day><month>12</month><year>2024</year></pub-date>
<pub-date pub-type="collection"><year>2024</year></pub-date>
<volume>11</volume><elocation-id>1504835</elocation-id>
<history>
<date date-type="received"><day>01</day><month>10</month><year>2024</year></date>
<date date-type="accepted"><day>04</day><month>11</month><year>2024</year></date>
</history>
<permissions>
<copyright-statement>&#x00A9; 2024 Xu, Zhong and Tan.</copyright-statement>
<copyright-year>2024</copyright-year><copyright-holder>Xu, Zhong and Tan</copyright-holder><license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by/4.0/">
<p>This is an open-access article distributed under the terms of the <ext-link ext-link-type="uri" xlink:href="http://creativecommons.org/licenses/by/4.0/">Creative Commons Attribution License (CC BY)</ext-link>. The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.</p></license>
</permissions>
<abstract>
<p>Coronary aneurysm, a dilated segment of the coronary artery, is a rare condition with a prevalence ranging from 0.02&#x0025; to 0.2&#x0025;. According to the current literature, reports of large aneurysms in the left main artery are extremely rare. We present a case of a 43-year-old male patient presenting with cough, wheezing, and dyspnea after a cold. Initial examinations suggested viral myocarditis, but further evaluation revealed a giant aneurysm in the left main coronary artery. Due to the high surgical risk, conservative treatment was chosen. Follow-up assessments showed no significant changes in the coronary aneurysm, with slight improvement in dyspnea. This rare case of a left main congenital coronary aneurysm suggests that treatment should consider the patient&#x0027;s overall condition, thrombosis presence, suitability for anticoagulant therapy, and aneurysm location and size.</p>
</abstract>
<kwd-group>
<kwd>coronary aneurysm</kwd>
<kwd>conservative treatment</kwd>
<kwd>coronary angiography</kwd>
<kwd>myocarditis</kwd>
<kwd>interventional therapy</kwd>
</kwd-group><counts>
<fig-count count="2"/>
<table-count count="0"/><equation-count count="0"/><ref-count count="14"/><page-count count="4"/><word-count count="0"/></counts><custom-meta-wrap><custom-meta><meta-name>section-at-acceptance</meta-name><meta-value>Coronary Artery Disease</meta-value></custom-meta></custom-meta-wrap>
</article-meta>
</front>
<body><sec id="s1" sec-type="background"><label>1</label><title>Background</title>
<p>Coronary angioma is characterized as a dilated segment of the coronary artery that exceeds 1.5 times the diameter of adjacent normal segments (<xref ref-type="bibr" rid="B1">1</xref>). Based on current literature, the prevalence of large coronary aneurysms ranges from 0.02&#x0025; to 0.2&#x0025; (<xref ref-type="bibr" rid="B2">2</xref>). However, the prevalence of large coronary aneurysms measuring 5&#x2005;cm or more in diameter is even lower, less than 0.02&#x0025; (<xref ref-type="bibr" rid="B1">1</xref>&#x2013;<xref ref-type="bibr" rid="B3">3</xref>). The right coronary artery is the most commonly affected, accounting for 40&#x0025;&#x2013;70&#x0025; of cases, followed by the circumferential artery (23&#x0025;) and the anterior descending branch (32&#x0025;) (<xref ref-type="bibr" rid="B1">1</xref>, <xref ref-type="bibr" rid="B2">2</xref>, <xref ref-type="bibr" rid="B4">4</xref>). Involvement of all three vessels or the left coronary trunk is rare.</p>
</sec>
<sec id="s2"><label>2</label><title>Case presentation</title>
<p>This case report presents the clinical details of a 43-year-old male patient who developed a cough and sneezing following a recent cold. Although the cold symptoms improved with self-administered medication, the patient experienced wheezing and paroxysmal dyspnea after physical activity. An electrocardiography(ECG) at a local hospital revealed rapid ventricular rate atrial fibrillation with variable conduction.</p>
<p>The cardiac ultrasound indicated that the LVEF was 38&#x0025; and the left atrium and ventricle were enlargement with weakened ventricular septal motion. Moderate mitral and mild aortic and pulmonary regurgitation were also noted. The patient also displayed a troponin sensitivity of 0.023&#x2005;ug/L. Based on these findings, the local hospital suspected viral myocarditis. The patient was referred to our cardiovascular department for further assessment. The patient&#x0027;s NT pro-BNP level was measured at 1,169&#x2005;pg/ml, and myocardial nuclide imaging (PET) revealed increased fibroblast activation protein inhibitors(FAPI) uptake throughout the left ventricle and right atrium (<xref ref-type="fig" rid="F1">Figure&#x00A0;1</xref>), supporting a diagnosis of myocarditis. The patient received symptomatic treatment, including hormone anti-inflammatory therapy, nutritional support for myocarditis, and gamma globulin therapy. However, these interventions provided limited relief from wheezing. Coronary angiography showed a left coronary dominant type, with no obvious stenosis in the left main trunk but a 2.7&#x2009;&#x00D7;&#x2009;2.3&#x2005;cm aneurysm in the distal part of the left main trunk. No stenosis was observed in the anterior descending branch, circumflex branch, or right coronary artery (<xref ref-type="fig" rid="F2">Figure&#x00A0;2</xref>). Autoimmune workup, including ANA, anti-dsDNA, ENA antibody profile, and ANCA, returned negative results, ruling out autoimmune causes of the aneurysm. Given the giant aneurysm in the left main trunk, myocarditis, and heart failure with intermediate ejection fraction, open-heart surgery with cardiopulmonary bypass, partial tumor resection, and coronary artery bypass grafting was recommended. However, due to the high risk, the patient and family opted for conservative treatment. As intravascular ultrasound (IVUS) was not performed during coronary angiography, antithrombotic or anticoagulant therapy was not initiated. Instead, the patient was prescribed atorvastatin 20&#x2005;mg nightly and metoprolol succinate sustained-release 47.5&#x2005;mg daily.</p>
<fig id="F1" position="float"><label>Figure 1</label>
<caption><p>Myocardial nuclide imaging (PET) revealed increased FAPI uptake throughout the left ventricle and right atrium.</p></caption>
<graphic xmlns:xlink="http://www.w3.org/1999/xlink" xlink:href="fcvm-11-1504835-g001.tif"/>
</fig>
<fig id="F2" position="float"><label>Figure 2</label>
<caption><p>Coronary angiography image and 3D reconstruction. <bold>(A)</bold> Coronary angiography showed a large aneurysm. <bold>(B,C)</bold> 3D reconstruction of an aneurysm in a coronary artery.</p></caption>
<graphic xmlns:xlink="http://www.w3.org/1999/xlink" xlink:href="fcvm-11-1504835-g002.tif"/>
</fig>
<p>The patient was closely monitored at 3, 6, 9, and 12 months post-discharge. During follow-up, the coronary aneurysm showed no significant changes, and the patient reported slight improvement in dyspnea, though other symptoms and physical signs remained stable.</p>
</sec>
<sec id="s3" sec-type="discussion"><label>3</label><title>Discussion</title>
<p>The exact cause of coronary aneurysm formation remains unclear. However, studies have shown that matrix metalloproteinases play a role in the pathogenesis of CAA formation by increasing the breakdown of proteins in the extracellular matrix (<xref ref-type="bibr" rid="B5">5</xref>, <xref ref-type="bibr" rid="B6">6</xref>). These enzymes have the ability to degrade various components of the arterial wall matrix and are found in higher concentrations in aneurysms. There is no consensus on the definition of a giant coronary aneurysm. In most literature, it is considered a giant coronary aneurysm if the dilation of the coronary artery is more than 1.5 times the diameter of adjacent normal reference vessels or if the vessel diameter is directly greater than 20&#x2005;mm (<xref ref-type="bibr" rid="B7">7</xref>&#x2013;<xref ref-type="bibr" rid="B9">9</xref>).</p>
<p>Coronary aneurysms can have several etiologies, including atherosclerosis, autoimmune diseases, and Kawasaki disease (<xref ref-type="bibr" rid="B9">9</xref>, <xref ref-type="bibr" rid="B10">10</xref>). In this case, the patient&#x0027;s autoimmune screening and coronary angiography findings were negative, suggesting a high likelihood of congenital origin. Coronary aneurysms can present with a range of clinical manifestations, such as asymptomatic chest pain, chest tightness, and pericardial tamponade (<xref ref-type="bibr" rid="B11">11</xref>). Initially, the patient did not experience any symptoms but was admitted to the hospital this time due to wheezing and dyspnea after catching a cold. Myocardial nuclide imaging was performed to consider myocarditis, but the symptoms did not improve significantly with treatment. Subsequent coronary angiography revealed the presence of a large coronary aneurysm.</p>
<p>Thrombus formation occurs in coronary aneurysms due to slow blood flow and platelet activation. To better assess aneurysm condition, intravascular ultrasound (IVUS) is recommended when a coronary aneurysm is detected via angiography. In the case with thrombosis, coagulation function, thromboelastography (TEG), and platelet function tests are advised. With timely anticoagulation or antithrombotic therapyare necessary. According to a 2021 study by Tuncay Taskesen, patients without symptoms, coronary occlusion, or thrombosis may not require specialized treatment (<xref ref-type="bibr" rid="B12">12</xref>). Additionally, Cihan Ozturk reported a case of an asymptomatic patient with a right coronary aneurysm who remained symptom-free for many years (<xref ref-type="bibr" rid="B13">13</xref>). However, for patients with obstructive coronary artery disease symptoms or myocardial ischemia due to embolization, surgical intervention is appropriate. Surgical options include interventional therapy, aneurysm excision, ligation, or coronary artery bypass surgery (<xref ref-type="bibr" rid="B2">2</xref>). In a notable 2024 case report by Najdat Bazarbashi, a patient with a large coronary aneurysm was successfully treated with a combination of stent placement and coil embolization (<xref ref-type="bibr" rid="B14">14</xref>). For cases involving multiple vessel disease, left main coronary artery aneurysm, or complications like fistula, compression, or rupture, surgical treatment is typically indicated. However, our patient opted for conservative management, which led to symptom alleviation over follow-up. If symptoms worsen, surgical or interventional treatments may become necessary, though surgical costs are high, and clinical evidence on specific prognostic outcomes is limited.</p>
</sec>
<sec id="s4" sec-type="conclusions"><label>4</label><title>Conclusion</title>
<p>Left main congenital coronary aneurysms are exceedingly rare. Proper diagnosis and treatment require assessment of thrombus presence within the aneurysm, along with evaluation of coagulation and platelet function to guide anticoagulation or antiplatelet therapy. For surgical decision-making, factors such as aneurysm location, size, arterial wall condition, and overall patient health should be carefully considered to select an appropriate treatment approach.</p>
</sec>
</body>
<back>
<sec id="s5" sec-type="data-availability"><title>Data availability statement</title>
<p>The original contributions presented in the study are included in the article/Supplementary Material, further inquiries can be directed to the corresponding author.</p>
</sec>
<sec id="s6" sec-type="ethics-statement"><title>Ethics statement</title>
<p>Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.</p>
</sec>
<sec id="s7" sec-type="author-contributions"><title>Author contributions</title>
<p>PX: Conceptualization, Writing &#x2013; original draft, Writing &#x2013; review &#x0026; editing. SZ: Data curation, Visualization, Writing &#x2013; review &#x0026; editing. HT: Writing &#x2013; original draft, Writing &#x2013; review &#x0026; editing.</p>
</sec>
<sec id="s8" sec-type="funding-information"><title>Funding</title>
<p>The author(s) declare that no financial support was received for the research, authorship, and/or publication of this article.</p>
</sec>
<sec id="s9" sec-type="COI-statement"><title>Conflict of interest</title>
<p>The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.</p>
</sec>
<sec id="s20" sec-type="ai-statement"><title>Generative AI statement</title>
<p>The author(s) declare that no Generative AI was used in the creation of this manuscript.</p>
</sec>
<sec id="s10" sec-type="disclaimer"><title>Publisher&#x0027;s note</title>
<p>All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.</p>
</sec>
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