• Li Xu, Yueqiu Su, and Zhou Leng.
• Operating Room, Department of Anesthesiology, West China Hospital, Sichuan University/West China School of Nursing, Sichuan University, Chengdu, Sichuan, China.
• Medicine (Baltimore). 2025 Jan 17; 104 (3): e41302e41302.

RationaleQuadricuspid aortic valve (QAV) is a rare condition with a very low incidence. Anomalous aortic origin of a coronary artery (AAOCA) is the most prevalent form of coronary anomaly. One variant of AAOCA is the anomalous aortic origin of the left coronary artery from the right coronary sinus (L-AAOCA). It is even rarer for QAV to occur in combination with AAOCA.Patient ConcernsIn this case report, we present a patient who was diagnosed with QAV and L-AAOCA using preoperative transesophageal echocardiography and 3-dimensional computed tomography (3D-CT).DiagnosesCardiac-enhanced CT showed an anomalous aortic origin of the left coronary artery from the right coronary sinus with an intramural course.InterventionsThe patient underwent sub-coronary ROSS procedure along with the unroofing procedure.OutcomesThe timely diagnosis of this incorporated cardiac anomaly, or rather its accidental discovery, led to successful surgical correction, eliminating the future risk of cardiac ischemia or SCD.LessonsThe timely diagnosis of this incorporated cardiac anomaly, or rather its accidental discovery, led to successful surgical correction, eliminating the future risk of cardiac ischemia or SCD.Copyright © 2025 the Author(s). Published by Wolters Kluwer Health, Inc.

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