Single coronary artery with right ventricular fistula: case report and literature review

Area of Science:

• Cardiology
• Pediatric Cardiology
• Congenital Heart Disease

Background:

• Coronary artery fistulae (CAF) are abnormal connections between a coronary artery and a heart chamber or vessel.
• A single coronary artery (SCA) is a rare congenital anomaly where only one coronary artery arises from the aorta.
• The coexistence of CAF and SCA is exceptionally rare, posing diagnostic and therapeutic challenges.

Observation:

• A 6-year-old boy presented with a diagnosed left single coronary artery and coronary artery fistulae communicating with the right ventricle.
• Preoperative angiography revealed a dilated, tortuous single coronary artery abnormally draining into the right ventricle.
• Intraoperative findings confirmed two distinct coronary artery fistulae entering the right ventricle.

Findings:

• Surgical ligation of the identified coronary artery fistulae was performed.
• Postoperative angiography demonstrated significant normalization of the single coronary artery's diameter, with residual mild tortuosity.
• The patient experienced an uncomplicated postoperative clinical course.

Implications:

• This case highlights the successful surgical management of a rare combination of congenital coronary anomalies.
• Effective treatment of coronary artery fistulae in the context of a single coronary artery can lead to favorable long-term outcomes.
• Reviewing similar reported cases aids in understanding and managing this complex pediatric cardiac condition.