scholarly journals Anomalous Left Coronary Artery Connected to the Pulmonary Artery in a 15-Year-Old Girl: Case Report and Discussion on Secondary Prevention of Sudden Death

2021 ◽  
Vol 2021 ◽  
pp. 1-6
Author(s):  
Jérémy Laïk ◽  
Virginie Fouilloux ◽  
Philippe Aldebert ◽  
Linda Koutbi ◽  
Jérôme Hourdain ◽  
...  

Background. Anomalous left coronary artery connected to the pulmonary artery (ALCAPA) is a rare congenital heart disease. Adaptive development of sufficient heterocoronary collaterality in the newborn may allow survival to a later age. In older children or adults, malignant ventricular arrhythmias can reveal the disease. Case Report. A 15-year-old girl was referred to the local hospital after a resuscitated out-of-hospital cardiac arrest. CT scan and coronary angiography revealed an ALCAPA. Direct aortic reimplantation of the left coronary artery was performed. Postoperative ECG monitoring showed short episodes of nonsustained ventricular tachycardia. Transthoracic echocardiography and cardiac MRI revealed subendocardial fibrosis of the anterolateral papillary muscle. Beta-blockade therapy was initiated at first intention. After hospital discharge, the patient reported several fainting without loss of consciousness. Considering sudden death nonrelated to effort, episodes of nonsustained ventricular tachycardia, and areas of myocardial fibrosis, the patient underwent subcutaneous cardioverter-defibrillator implantation. 6-month follow-up is satisfactory without clinical or rhythmic abnormalities. Discussion. Indication for surgical correction of ALCAPA is well defined, but rhythmic secondary prevention after resuscitated cardiac arrest is less consensual. Cardiac MRI is an essential tool in the identification of a potential rhythmic substrate and should be taken into account in the discussion of a preventive cardioverter-defibrillator implantation.

2020 ◽  
Vol 4 (3) ◽  
pp. 1-5
Author(s):  
Madelien V Regeer ◽  
Olga Bondarenko ◽  
Katja Zeppenfeld ◽  
Anastasia D Egorova

Abstract Background Anomalous left coronary artery from the pulmonary artery (ALCAPA) is a rare congenital disorder resulting in ischaemia and myocardial infarction which can act as a potential substrate for life-threatening arrhythmias and sudden cardiac death. Case summary A 19-year-old man was admitted to the hospital after successful resuscitation from an out-of-hospital cardiac arrest (OHCA) due to ventricular fibrillation occurring during jogging. In the diagnostic work-up of the OHCA, computed tomography identified an ALCAPA. The patient was referred to our tertiary hospital for surgical correction. Direct reimplantation of the left coronary artery in the aorta was performed. During follow-up, 24-h electrocardiogram revealed short episodes of non-sustained ventricular tachycardia (VT). The magnetic resonance imaging at initial admission showed focal wall thinning and transmural late gadolinium enhancement consistent with a previous anterolateral myocardial infarction. Therefore, the aetiology of the OHCA could be due to a scar-related mechanism and not necessarily due to a reversible cause and an implantable cardioverter-defibrillator (ICD) was considered indicated. Given the young age and the lower complication rates, a subcutaneous device was preferred over a transvenous ICD. However, as a subcutaneous ICD (S-ICD) lacks the possibility of anti-tachycardia pacing, programmed electrical stimulation (PES) was performed to test for inducibility of monomorphic, re-entrant VT. After a negative PES, an S-ICD was implanted. Discussion ALCAPA is a potential cause of OHCA in young patients. Some of these patients keep an irreversible substrate for ventricular arrhythmias despite full surgical revascularization and might be candidates for (subcutaneous) ICD implantation.


Author(s):  
Yew Eng Tan ◽  
Kok King Chia ◽  
Noor Khairiah Abdul Karim

Abstract Background Anomalous left coronary artery from the pulmonary artery (ALCAPA) is a rare congenital anomaly where the left main coronary artery arises from the pulmonary artery, instead of the coronary sinus of the ascending aorta. ALCAPA is divided into infant and adult types. Life-threatening complications such as malignant arrhythmia and sudden death could ensue in adult type of ALCAPA. Imaging is the current preferred modality for ALCAPA. It is indeed a challenge to diagnose ALCAPA due to its non-specific clinical presentation and laboratory findings. We report a case of adult type ALCAPA presented with unstable angina surprisingly surviving into adulthood with no symptoms prior to presentation, which is extremely rare. Case presentation A 53-year-old lady presented with sudden onset of chest pain and worsening shortness of breath. She was initially treated as unstable angina. Physical examination revealed normal heart sound. Computed tomography coronary angiography (CTCA) showed an anomalous origin of the left coronary artery from the posterior wall of the proximal pulmonary artery, compatible with ALCAPA. The anomalous left coronary artery bifurcates into left anterior descending and left circumflex arteries. Cardiothoracic surgeon planned for occlusion of ALCAPA via the pulmonary artery, owing to the fact that unfeasible rerouting in the presence of well-established collateral supply. Conclusions ALCAPA is a rare and life-threatening condition in adults which may lead to myocardial infarction and sudden death in untreated cases. CTCA is one of the preferred modern imaging modality in ALCAPA owing to its superior ability for direct visualization of the anomaly. Hence, early identification and surgical intervention of the anomaly are paramount to reduce the morbidity and mortality.


2020 ◽  
Vol 10 (2) ◽  
pp. 25-32
Author(s):  
Mita Singh ◽  
Ana Teresa Gomes ◽  
Moad El-Haddad ◽  
Abdel Khalid Saidmeerasah ◽  
Rashid Iqbal

Anomalous Left Coronary Artery from the Pulmonary Artery (ALCAPA) is a rare coronary artery anomaly which accounts for 0.25–0.5% of all congenital cardiac diseases, where most die within the first year of life. We present a case report of a 50-year-old lady who presented to hospital with persistent palpitations. Her admission electrocardiogram found her to be in Atrial Fibrillation (AF). She was rate-controlled and subsequently discharged. Despite that, she represented with further episodes of AF and was referred for an outpatient transthoracic echocardiogram. This revealed a dilated right coronary artery, retrograde flow in the left coronary artery and collateral flow in the myocardium. To investigate, the patient had undergone further imaging which confirmed the diagnosis. As such, she was later shortlisted for surgical intervention. Conclusively, our case exemplifies the role of multimodal imaging to identify the features of ALCAPA and may be useful for the purposes of surgical intervention.


2007 ◽  
Vol 10 (2) ◽  
pp. 142 ◽  
Author(s):  
Gaurab Maitra ◽  
Saibal Roychowdhury ◽  
Somnath Ganguli ◽  
Susmit Bhattacharya ◽  
Ajay Kaul

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