A case of anomalous origin of the left coronary artery from the pulmonary artery presenting with sudden cardiac arrest due to coronary artery steal generated by excessive exercise

Imamura, Takaaki; Kondo, Hidekazu; Ishii, Yumi; Haraguchi, Miho; Kobukata, Mami; Otsubo, Toyokazu; Saito, Shotaro; Akioka, Hidefumi; Shinohara, Tetsuji; Yufu, Kunio; Takahashi, Naohiko

doi:10.1016/j.jccase.2016.07.001

Cited by 2 publications

(6 citation statements)

References 7 publications

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“…The pathophysiology and hence, the clinical presentation go through 3 phases. [3] Initially, due to the persistent high pulmonary artery pressure, blood from the pulmonary artery flows into the anomalous left coronary artery is maintained, and the infant is asymptomatic. During the next phase, with the closure of the ductus arteriosus and the decreasing pulmonary artery pressure, the blood flow through the anomalous artery becomes increasingly compromised and eventually becomes retrograde.…”

Section: Discussionmentioning

confidence: 99%

“…[2–6] In patients with ALCAPA syndromes, only 10% survive beyond childhood. [3] The occurrence of this anomaly is not confined to the pediatric population as it can, in sporadic cases, manifest in adults with catastrophic presentation as in this autopsy. When clinically asymptomatic, these anomalies are discovered incidentally during the angiogram, a computed tomography (CT) or echocardiography.…”

Section: Introductionmentioning

confidence: 89%

“…When clinically asymptomatic, these anomalies are discovered incidentally during the angiogram, a computed tomography (CT) or echocardiography. [3,7,8]…”

Section: Introductionmentioning

confidence: 99%

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Anomalous origin of left coronary arteries from the pulmonary artery in an asymptomatic adult

Shaarani

AlZubaidi²,

Jin³

2019

Medicine

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Rationale:Anomalous Origin of Left Coronary Arteries from the Pulmonary Artery (ALCAPA), also known as Bland-White-Garland (BWG), is a rare form of coronary artery anomaly that is usually discovered in the first few months of life. Only rarely can patients with this anomaly reach adulthood without symptoms.Patient concerns:A 28-year-old female was witnessed suddenly collapse with a seizure-like episode by her colleagues at work.Diagnosis and Intervention:Routine cardiopulmonary resuscitation was performed by emergency medical service technologists. The patient was unable to be revived. Postmortem examination revealed the patient had ALCAPA with a focal chronic ischemic injury of the left ventricle. Moreover, a high take-off of the right coronary artery was also discovered.Outcomes:The patient passed away due to ALCAPA. The mechanism of death was cardiac arrhythmia being triggered by myocardial ischemic changes.Lessons:In the rare cases where ALCAPA manifests in an asymptomatic adult, the mortality rate is very high. This case demonstrates the importance of awareness of such patients living under the tremendous risk of sudden cardiac death.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 89%

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Anomalous origin of left coronary arteries from the pulmonary artery in an asymptomatic adult

Shaarani

AlZubaidi²,

Jin³

2019

Medicine

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“…La malformación de las arterias coronarias corresponde a un grupo infrecuente de anomalías congénitas de las que la más conocida es la fístula coronaria, que genera isquemia miocárdica por robo de flujo coronario (1) . Debido a que este grupo constituye una entidad congénita infrecuente, que afecta a menos del 1% de la población (2) , se desconocen datos fidedignos de su incidencia.…”

Section: Discussionunclassified

“…Esta entidad reviste especial importancia por su alta mortalidad en edades tempranas. La supervivencia en la infancia es cercana al 10% (4) , y los que logran sobrevivir pueden presentar muerte súbita durante la juventud (1,4,5) . La presentación clínica más agresiva en edades tempranas se debería a la ausencia de circulación colateral que ayudaría a mantener la perfusión miocárdica en adultos (3) .…”

Section: Discussionunclassified

Nacimiento anómalo de la arteria coronaria izquierda desde el tronco de la arteria pulmonar con isquemia miocárdica e insuficiencia mitral

Oliveri

Chango-Azanza

Deviggiano

et al. 2019

Rev Ecocar Pract (RETIC)

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Presentamos el caso de una paciente con diagnóstico de nacimiento anómalo de la arteria coronaria izquierda desde el tronco de la arteria pulmonar (ALCAPA). Se trata de una paciente joven con diagnóstico de insuficiencia mitral severa asintomática de causa no esclarecida. La inesperada evidencia de isquemia en territorio anterior y apical en el ecocardiograma de estrés solicitado para evaluación funcional sumado a los signos característicos del ecocardiograma sugirió el diagnóstico presuntivo de ALCAPA, que se confirmó con angiotomografía coronaria. Entre los pocos pacientes con ALCAPA que alcanzan la vida adulta, la isquemia miocárdica como presentación inicial es poco frecuente.

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A case of anomalous origin of the left coronary artery from the pulmonary artery presenting with sudden cardiac arrest due to coronary artery steal generated by excessive exercise

Cited by 2 publications

References 7 publications

Anomalous origin of left coronary arteries from the pulmonary artery in an asymptomatic adult

Anomalous origin of left coronary arteries from the pulmonary artery in an asymptomatic adult

Nacimiento anómalo de la arteria coronaria izquierda desde el tronco de la arteria pulmonar con isquemia miocárdica e insuficiencia mitral

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