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Fetal Isolated Anomalous Origin of Right Pulmonary Artery from Aorta.

ABSTRACT: The anomalous origin of a branch pulmonary artery from the aorta (AOPA) is characterized by the anomalous origin of one of the branch pulmonary arteries (PA) from the ascending aorta and a normal origin of the other PA from main PA. AOPA is an extremely rare cardiac malformation. Few studies have reported fetal anomalous origin of PA from aorta with other malformation. We report a case of isolated distal anomalous origin of the right PA from the aorta that was diagnosed by fetal echocardiography at 25 weeks' of gestation. Tracing the course of PA branches is important to make diagnosis.

SUBMITTER: Zeng S

PROVIDER: S-EPMC4502634 | biostudies-other | 2015 Apr

REPOSITORIES: biostudies-other

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Project description:Anomalous right coronary artery from pulmonary artery (ARCAPA) is one of four known anomalies of the coronary arteries originating from the pulmonary artery. ARCAPA is a rare congenital anomaly that is associated with possible long-term complications including myocardial ischemia and left ventricular dysfunction. Clinical presentation can vary from asymptomatic murmur to angina, dyspnea, fatigue, congestive heart failure, myocardial infarction and even cardiac arrest. We present a case of a 52-year-old male who presented to the hospital for intermittent chest pain. The patient was vitally stable and examination was unremarkable. Upon workup, electrocardiogram (EKG) on admission showed no acute ischemic changes and troponin were <0.03 ng/mL. The patient was evaluated by cardiology and had a dobutamine stress echocardiogram (ECHO) for ischemic evaluation. During the recovery phase of dobutamine stress ECHO, the patient reported having 10/10 chest pain with a drop in blood pressure to 90 mmHg systolic. He was subsequently given sublingual nitroglycerin and fluids. Pain did resolve and blood pressure improved after treatment. No wall motion abnormalities were noted on ECHO, but there were inferior ST depressions on EKG during the stress portion of the test. The patient had repeated blood work after the stress test which showed an elevated troponin level of 0.08 ng/mL. The patient was taken to the catheterization lab for diagnostic coronary angiogram. Cardiac catheterization showed a right coronary artery (RCA) arising from the pulmonary artery and diagnosis of isolated ARCAPA was made because of his intermittent chest pain. No intervention was performed as no significant lesions were found. The patient was started on medical therapy including aspirin, beta blocker and statin. The patient did not want to pursue surgery at that point and he did not want any further intervention. He was discharged home with instructions to follow up with cardiology in an outpatient setting to be referred for surgical repair. The purpose of writing this case is to increase awareness among physicians to be able to recognize possible anomalous coronary artery origins during cardiac catheterization and to understand the risk of long-term complications of ARCAPA and need for surgical repair.

Project description:A 58-year-old female visited our hospital complaining of fatigue and repetitive pre-syncope during exercise. She had suffered with those symptoms for decades and had visited some hospitals where she did not get conclusive diagnosis. She was ultimately diagnosed as having subclinical ventricular septal defect unrelated to her symptoms. Echocardiography revealed normal ventricular function, but color Doppler showed multiple abnormal intramyocardial blood flow signals in ventricular septum and moderator band, which suggested abnormally developed coronary perfusion. Dilated left main trunk and abnormal color Doppler signal running into the main pulmonary trunk were also observed. Coronary computed tomography angiography revealed that right coronary artery originated from pulmonary artery and made connection to distal left anterior descending artery, which led to the final diagnosis of anomalous origin of the right coronary artery from the pulmonary artery. Exercise stress echocardiography showed wall motion abnormalities in inferior region which suggested ischemia in right coronary artery (RCA) territory. Her symptoms could be derived from the myocardial ischemia. She was referred to surgical intervention considering the possible risk of cardiac events including sudden death. Re-implantation of the abnormal RCA to the aorta was performed successfully. Her long-year persistent symptoms were completely alleviated, and repeat exercise stress echocardiography showed no evidence of ischemia. <Learning objective: We report an adult case of symptomatic anomalous origin of the right coronary artery from the pulmonary artery, who had not been given the correct diagnosis. Noninvasive diagnostic examinations including transthoracic Doppler echocardiography, exercise stress echocardiography and coronary computed tomography angiography contributed to the anatomical and physiological assessment of the rare congenital heart disease before and after the successful coronary re-implantation surgery.>.

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Fetal Isolated Anomalous Origin of Right Pulmonary Artery from Aorta.

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