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Eur Heart J Case Rep
Eur Heart J Case Rep
ehjcr
European Heart Journal. Case Reports
2514-2119
Oxford University Press UK

10.1093/ehjcr/ytae468
ytae468
Images in Cardiology
AcademicSubjects/MED00200
Eurheartj/31
Eurheartj/32
An adult case of Bland–White–Garland syndrome with Vieussens’ arterial ring
https://orcid.org/0000-0003-0176-542X
Hagiwara Hikaru Department of Cardiovascular Medicine, Kushiro City General Hospital, 1-12, Shunkodai, Kushiro 085-0822, Japan
Department of Cardiovascular Medicine, Faculty of Medicine and Graduate School of Medicine, Hokkaido University, Kita-15, Nishi-7, Kita-ku, Sapporo 060-8638, Japan

Komoriyama Hirokazu Department of Cardiovascular Medicine, Kushiro City General Hospital, 1-12, Shunkodai, Kushiro 085-0822, Japan

Kato Yoshiya Department of Cardiovascular Medicine, Kushiro City General Hospital, 1-12, Shunkodai, Kushiro 085-0822, Japan
Department of Cardiovascular Medicine, Faculty of Medicine and Graduate School of Medicine, Hokkaido University, Kita-15, Nishi-7, Kita-ku, Sapporo 060-8638, Japan

Anzai Toshihisa Department of Cardiovascular Medicine, Faculty of Medicine and Graduate School of Medicine, Hokkaido University, Kita-15, Nishi-7, Kita-ku, Sapporo 060-8638, Japan

Bordignon Stefano Handling Editor
Canpolat Ugur Editor
Corresponding author. Tel: +81-11-706-6973, Email: hikaruh731@med.hokudai.ac.jp
Conflict of interest: None declared.

9 2024
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© The Author(s) 2024. Published by Oxford University Press on behalf of the European Society of Cardiology.
2024
https://creativecommons.org/licenses/by/4.0/ This is an Open Access article distributed under the terms of the Creative Commons Attribution License (https://creativecommons.org/licenses/by/4.0/), which permits unrestricted reuse, distribution, and reproduction in any medium, provided the original work is properly cited.
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pmcCase description

Bland–White–Garland (BWG) syndrome is a rare congenital cardiac anomaly for which only a few patients survive into adulthood.1 This report presents the rare case of a 58-year-old patient with BWG syndrome who did not receive surgical treatment for the condition.

A 58-year-old woman visited a local clinic complaining of shoulder pain and found to have poor R-wave progression electrocardiogram (Figure 1A) and was referred to our hospital. Echocardiogram showed anterior wall motion abnormality and decreased left ventricular ejection fraction of 42%. Coronary angiography revealed right coronary artery dominance (see Supplementary material online, Video S1) and no visible left main stem artery (see Supplementary material online, Video S2). Furthermore, a connection was observed between the conus branch and the left coronary and pulmonary arteries (see Supplementary material online, Videos S3 and S4). Volume-rendered computed tomography showed a Vieussens’ arterial ring (Figure 1B and C), a connection between the conus artery and the diagonal branches of the left anterior descending coronary artery, and an anomalous origin of the left coronary artery from the pulmonary artery (Figure 1D and E).

Figure 1 Twelve-lead electrocardiography (A) and volume-rendered computed tomography of the heart of a 58-year-old patient with Bland–White–Garland syndrome showing complex anomalous vessels. The conus artery arises from the right sinus of Valsalva and connects to the proximal portion of the left anterior descending coronary artery (B, C). The left coronary artery anomalously originates from the pulmonary artery (D, E). Ao, aorta; RCA, right coronary artery; RCC, right coronary cusp; LAD, left anterior descending artery; PA, pulmonary artery.

Patients with BWG syndrome can die abruptly; ∼90% of them die at an average age of 35 years.1 The present case had rich collateral circulation from the conus artery, which increased blood flow in the left coronary artery and reduced the degree of myocardial ischaemia, enabling her to live up to 58 years of age. Previous reports have suggested that patients with BWG syndrome should undergo surgical intervention, regardless of the presence or absence of clinical symptoms or complications, to prevent the development of heart failure and reduce the risk of sudden cardiac death; therefore, surgical treatment was planned in this case.1

Supplementary Material

ytae468_Supplementary_Data

Supplementary material

Supplementary material is available at European Heart Journal – Case Reports online.

Consent: Informed consent was provided by the patient.

Funding: None declared.

Data availability

Data sharing is not applicable as no data sets were generated or analysed for this case report.
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Reference

1 Luo H , KwakuOE, LaiY, YueR. Adult-type anomalous origin of the left coronary artery from the pulmonary artery and right coronary-right atrial fistula: a case report. BMC Cardiovasc Disord 2024;24 :31.38183012
