Medicine · 2025
Anomalous left coronary artery from the pulmonary artery: A case report of ALCAPA syndrome with bicuspid aortic valve.
СтатьяКлинический случайНазвание и аннотация: Перевод в очереди
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Jabareen Maaweya, Alhroub Wasef, Nassr Mohammed, Nassar Hisham, Emar Ahmad, Alhusaini Zaid, Hasasna Ahmad
Аннотация
RATIONALE: Anomalous left coronary artery from the pulmonary artery (ALCAPA) syndrome, also known as Bland-White-Garland syndrome, is a rare congenital heart defect predominantly diagnosed in infants and associated with high early mortality rates without surgical intervention. While ALCAPA generally presents alone, it can sometimes occur alongside other congenital anomalies, such as a bicuspid aortic valve (BAV), a combination scarcely documented in the literature. This case report explores a rare presentation of ALCAPA syndrome in a 27-year-old adult with concurrent BAV.
PATIENT CONCERNS: The patient, a 27-year-old male, presented with progressive shortness of breath over several months, without additional symptoms such as chest pain or palpitations. His medical history was unremarkable, and he had no prior cardiovascular issues or significant family history of sudden cardiac death.
DIAGNOSES: A physical examination revealed stable vital signs and an early diastolic murmur. Further investigations, including echocardiography and computed tomography coronary angiography, confirmed the diagnosis of ALCAPA syndrome with a BAV and mild to moderate aortic regurgitation. Cardiac catheterization demonstrated sufficient collateral circulation from the right coronary artery to the left coronary system, permitting adequate myocardial perfusion.
INTERVENTIONS: Due to the presence of adequate collateral circulation, the decision was made to manage the patient medically instead of surgically. The patient was started on a regimen of rivaroxaban, bisoprolol, and ramipril to manage his condition and reduce the risk of thrombus formation, in addition to providing myocardial protection.
OUTCOMES: The patient responded positively to medical management, remaining asymptomatic with stable cardiovascular function at follow-up. After 2 years of follow-up, the patient continued to do well, with no reported symptoms.
LESSONS: This case emphasizes the importance of recognizing ALCAPA in adults, particularly in patients presenting with minimal symptoms despite significant congenital anomalies. In select cases with adequate collateral circulation, medical management may be a viable alternative to surgical intervention, underscoring the need for individualized treatment approaches based on the patient's anatomical and functional profile.
PATIENT CONCERNS: The patient, a 27-year-old male, presented with progressive shortness of breath over several months, without additional symptoms such as chest pain or palpitations. His medical history was unremarkable, and he had no prior cardiovascular issues or significant family history of sudden cardiac death.
DIAGNOSES: A physical examination revealed stable vital signs and an early diastolic murmur. Further investigations, including echocardiography and computed tomography coronary angiography, confirmed the diagnosis of ALCAPA syndrome with a BAV and mild to moderate aortic regurgitation. Cardiac catheterization demonstrated sufficient collateral circulation from the right coronary artery to the left coronary system, permitting adequate myocardial perfusion.
INTERVENTIONS: Due to the presence of adequate collateral circulation, the decision was made to manage the patient medically instead of surgically. The patient was started on a regimen of rivaroxaban, bisoprolol, and ramipril to manage his condition and reduce the risk of thrombus formation, in addition to providing myocardial protection.
OUTCOMES: The patient responded positively to medical management, remaining asymptomatic with stable cardiovascular function at follow-up. After 2 years of follow-up, the patient continued to do well, with no reported symptoms.
LESSONS: This case emphasizes the importance of recognizing ALCAPA in adults, particularly in patients presenting with minimal symptoms despite significant congenital anomalies. In select cases with adequate collateral circulation, medical management may be a viable alternative to surgical intervention, underscoring the need for individualized treatment approaches based on the patient's anatomical and functional profile.