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A Middle-Aged Man With Pulseless VT and Dual Pathology: Anomalous Left Main Coronary Artery From Right Coronary Cusp

Amir Heidari1, Enssieh Hashemi2, Mehrdad Jafari Fesharaki3

  • 1Department of Cardiology, Imam Hossein Hospital Shahid Beheshti University of Medical Sciences Tehran Iran.

Clinical Case Reports
|March 20, 2026
PubMed

Insights

Anomalous aortic origin of a coronary artery from the opposite sinus (AOCAOS) with a transseptal course is rare. This case highlights its potential severity when coexisting with dilated cardiomyopathy, emphasizing comprehensive diagnostics.

Area of Science:

  • Cardiology
  • Cardiovascular Imaging
  • Medical Case Reports

Background:

  • Anomalous aortic origin of a coronary artery from the opposite sinus (AOCAOS) is a rare congenital heart defect.
  • A left main coronary artery (LMCA) arising from the right coronary cusp (RCC) with a transseptal course is an uncommon and potentially serious variant.
  • The co-occurrence of AOCAOS and dilated cardiomyopathy (DCM) is exceptionally rare, posing significant diagnostic and therapeutic challenges.

Purpose of the Study:

  • To present a rare case of anomalous aortic origin of a coronary artery from the opposite sinus (AOCAOS) with a transseptal course coexisting with dilated cardiomyopathy (DCM).
  • To highlight the diagnostic utility of advanced imaging modalities and the clinical management of such a complex case.
  • To underscore the potential severity of transseptal AOCAOS when associated with cardiomyopathy.

Main Methods:

  • Case report of a 43-year-old male resuscitated from ventricular tachycardia.
  • Diagnostic workup included echocardiography, invasive coronary angiography, cardiac magnetic resonance (CMR), and coronary computed tomography angiography (CCTA).
  • Medical management for heart failure and implantation of a cardiac resynchronization therapy defibrillator (CRT-D) for primary prevention of sudden cardiac death.

Main Results:

  • The patient presented with severely reduced left ventricular ejection fraction (LVEF) of 20% and was diagnosed with DCM.
  • Coronary angiography revealed anomalous origin of the LMCA from the RCC.
  • CCTA delineated a transseptal course of the LMCA, and CMR confirmed DCM without fibrosis. Significant clinical improvement was noted at 3-month follow-up.

Conclusions:

  • Transseptal AOCAOS, particularly when associated with DCM, may not be benign and can present challenges in patients with sudden cardiac death.
  • Comprehensive diagnostic evaluation, including CCTA and CMR, is crucial for complete assessment of complex coronary anomalies.
  • Multimodality imaging and appropriate device therapy are essential in managing patients with this rare combination of conditions.

Keywords:
anomalous left main coronary arterycardiac arrestdilated cardiomyopathytransseptal courseventricular tachycardia

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