Anomalous origin of the left coronary artery from the pulmonary artery presenting as dilated cardiomyopathy: a case report

Gribaa, Rym; Slim, Mehdi; Salem, Helmi Ben; Neffati, E.; Boughzéla, Essia

doi:10.1186/1752-1947-8-170

Cited by 13 publications

(12 citation statements)

References 12 publications

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“…Thus, symptomatic infants often have a severe clinical course and warrant prompt surgery once diagnosis is established. LV dilation and anterior leaflet prolapse of the mitral valve are the most readily discernable signs of echocardiography; meanwhile, the clinical sign is apical systolic murmur, but could lead to a false diagnosis of dilated cardiomyopathy (16) or congenital mitral valve prolapse. In this situation, dilated RCA and increased RCA/ AO ratio were the helpful signs in distinguishing ALCAPA from dilated cardiomyopathy; echogenic chordae tendineae or the papillary muscle of the mitral valve also indicates an ischemic origin and provides a valuable clue for ALCAPA detection (17).…”

Section: Discussionmentioning

confidence: 99%

Diagnostic value of echocardiography on detecting the various types of anomalous origin of the left coronary artery from the pulmonary artery

Yu¹,

Wang²,

Wang³

et al. 2020

J Thorac Dis

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Background: To assess the diagnostic value of echocardiography in detecting the various types of anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA). Methods: A total of 30 patients with an established diagnosis of ALCAPA were retrospectively analyzed, and classified into infant-(n=20) and adult-type (n=10) groups according to the age of symptom manifestation and the mode of presentation. All patients underwent echocardiography examination. Results: Twenty-four out of thirty patients were diagnosed with ALCAPA by echocardiography. The remaining six cases were confirmed by dual-source computed tomography (DSCT) and angiocardiography, respectively. In the infant-type group, there was negligible or no collateral flow between the right coronary artery (RCA) and the left coronary artery (LCA). Eighteen of these patients had enhanced echogenicity of left ventricular (LV) papillary muscles, different degrees of mitral regurgitation (MR) and the RCA to aortic annulus ratio (RCA/AO) was >0.12. In the adult-type group, all ten patients had RCA dilation and significant development of collateralization from the RCA to the dilated LCA. They all had mild MR and RCA/AO was >0.20. Preoperatively, left ventricular ejection fraction (LVEF) was significantly lower in infant-type group than in adult-type group (46.24%±5.47% vs. 61.43%±6.38%, P<0.01). Cardiac surgery significantly improved post-operative LVEF (60.12%±6.02%, P<0.01 vs. pre-operation) in infant-type group. Conclusions: Echocardiography plays a pivotal role in detecting ALCAPA. Imaging and clinical features differ significantly between infant-and adult-type cases.

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Section: Discussionmentioning

confidence: 99%

Diagnostic value of echocardiography on detecting the various types of anomalous origin of the left coronary artery from the pulmonary artery

Yu¹,

Wang²,

Wang³

et al. 2020

J Thorac Dis

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“…La coronaire gauche naissant de l'artère pulmonaire va vasculariser le myocarde ventriculaire gauche par du sang pauvre en oxygène susceptible d'induire des lésions d'ischémie à l'effort ou bien elle est perfusée de façon rétrograde à partir de la coronaire droite et se vide dans l'artère pulmonaire lorsque les pressions pulmonaires baissent, entrainant un vol coronaire [6]. Plusieurs auteurs ont déjà rapporté des cas de cette pathologie, notamment des cas similaires au notre [7,8]. La particularité de notre cas est que c'est le seul et le premier à être rapporté à Madagascar.…”

Section: Commentaireunclassified

Naissance Anormale Du Tronc Coronaire Gauche A Partir De L’artere Pulmonaire : à propos d’un cas (Anomalous Left Coronary Artery From Pulmonary Artery: about a case)

Ravaoavy¹,

Elsa²,

Ritchy³

et al. 2019

CMRO

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Anomalous left coronary artery from pulmonary artery ALCAPA, is a rare congenital heart disease. It affects one child in 300,000 births. It causes myocardial ischemia leading to impaired left ventricle systolic function and signs of left heart failure. Several authors have already reported cases of ALCAPA. We are reporting for the first time a case seen in Madagascar. This is a Franco-Malagasy child aged 4 months and 3 weeks, hospitalized at the Mother and Child University Hospital of Tsaralalana for progressively worsening dyspnea. At entry, the infant presented with cyanosis and signs of cardiogenic shock: hypotension, oligoanuria and cold extremities. The physical examination essentially found pulmonary rales, a 2/6 systolic murmur of mitral insufficiency and hepatomegaly. Chest X-ray showed significant cardiomegaly. The electrocardiogram found a Q wave of necrosis in DI, AVL and V2 to V6. The echocardiogram coupled with Doppler performed at the Soavinandriana Hospital concluded that the left coronary artery was connected to the pulmonary artery. The progression after 6 days in hospital was marked by the disappearance of signs of shock and dyspnea after treatment with Dobutamine, diuretics and Salbutamol. The child was proposed for a surgical cure, the gold standard of treatment for ALCAPA. Resume: La naissance anormale du tronc coronaire gauche à partir de l'artère pulmonaire ou ALCAPA, de l'anglais anomalous left coronary artery from pulmonary artery ou encore syndrome de Bland-White-Garland, est une pathologie congénitale rare. Elle affecte un enfant sur 300 000 naissances. Elle entraine une ischémie myocardique à l'origine d'une altération de la fonction systolique du ventricule gauche et des signes d'insuffisance cardiaque gauche. Plusieurs auteurs ont déjà rapporté les cas de AL-CAPA. Nous rapportons pour la première fois un cas vu à Madagascar. Il s'agit d'un nourrisson Franco-Malgache de 4 mois et 3 semaines, hospitalisé au Centre Hospitalier Universitaire Mère-Enfant de Tsaralalana pour une dyspnée d'aggravation progressive. A l'entrée, le nourrisson présentait une altération de l'état général, cyanose et des signes de choc cardiogénique : hypotension artérielle, oligoanurie et extrémités froides. L'examen physique retrouvait essentiellement des râles crépitants diffus, un souffle systolique 2/6 d'insuffisance mitrale et une hépatomégalie. La radiographie du thorax de face montrait une cardiomégalie importante. L'électrocardiogramme a retrouvé une onde Q de nécrose en DI, AVL et de V2 jusqu'en V6. L'échocardiogramme couplé au Doppler réalisé au Centre Hospitalier de Soavinandriana a conclu à l'anomalie de connexion du tronc de l'artère coronaire gauche à partir de l'artère pulmonaire. L'évolution après 6 jours d'hospitalisation a été marquée par la disparition des signes de choc et de la dyspnée après un traitement par Dobutamine, diurétiques et Salbutamol. L'enfant a été proposé pour une cure chirurgicale, le gold standard du traitement de l'ALCAPA.

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“…Due to atypical signs and symptoms in childhood, it can be misdiagnosed as dilated cardiomyopathy [ 54 ] . The definitive diagnosis can be reached by multislice computed tomography and coronary angiography [ 55 ] . Conventional angiography and magnetic resonance imaging depict the origin and proximal courses of these arteries [ 56 ] .…”

Section: Diagnosismentioning

confidence: 99%

Anomalous origin of coronary artery: taxonomy and clinical implication

Yuan¹

2014

Revista Brasileira De Cirurgia Cardiovascular

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ObjectiveAnomalous origin of coronary artery is uncommon. The taxonomies of anomalous origin of coronary artery are inconsistent and complex. Conceptual and therapeutic debates remain. The aim of the present study is to reappraise the concept of anomalous origin of coronary artery and to discuss the potential hazards and treatment rationale of this anomaly on basis of literature review.MethodsA comprehensive literature review was made in terms of the taxonomies including “simple”, “multiple” and “complex” types of anomalous origin of coronary artery.ResultsAnomalous origin of coronary artery can be simply categorized according to the ectopically originated coronary artery. There are a couple of complex anatomical variants: “multiple” type, involving more than one coronary artery or branch, which can be subdivided into 2 subtypes, A) more than one coronary arteries or branches arising from one place; and B) two coronary arteries/branches arising from separate ectopic sites; and “complex” type, associated with acquired heart disease, or congenital heart defects.ConclusionSudden cardiac death in anomalous origin of coronary artery is associated with the anatomical features including abnormal coursing, acute angle take-off and ostial abnormalities. Atherosclerosis is prone to be in the right-sided ectopic and retroaortic coursing coronary artery. Surgical treatment is a definitive therapy. Simple coronary artery bypass grafting is not recommended due to the potential hazards of coronary steal phenomenon and poor patency of mammary arterial grafts, and modified maneuvers such as coronary ostial reimplantation, impinged coronary segment unroofing and coronary stent deployment are advocated instead.

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Anomalous origin of the left coronary artery from the pulmonary artery presenting as dilated cardiomyopathy: a case report

Cited by 13 publications

References 12 publications

Diagnostic value of echocardiography on detecting the various types of anomalous origin of the left coronary artery from the pulmonary artery

Diagnostic value of echocardiography on detecting the various types of anomalous origin of the left coronary artery from the pulmonary artery

Naissance Anormale Du Tronc Coronaire Gauche A Partir De L’artere Pulmonaire : à propos d’un cas (Anomalous Left Coronary Artery From Pulmonary Artery: about a case)

Anomalous origin of coronary artery: taxonomy and clinical implication

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