Magnetic resonance studies in management of adult cases with Bland-White-Garland syndrome

Komócsi, András; Símor, Tamás; Tóth, Levente; Szabados, S.; Mágel, Ferenc; Pintér, Tünde; Aradi, Dániel; Kónyi, Attila; Horváth, Iván; Papp, L.

doi:10.1016/j.ijcard.2006.11.084

Cited by 16 publications

(11 citation statements)

References 5 publications

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“…Given the classic concept of “adult-type” of ALCAPA [26,27], intercoronary collateralization allows these patients to reach adulthood and protects the myocardium from irreversible damage, which, in turn, raises the question whether surgical repair is required in adulthood. Indeed, Komocsi and colleagues reported three adult cases with uncorrected ALCAPA and found only moderate ischemia and limited necrosis in two of the three patients that had normal or only mildly impaired LV function [28]. …”

Section: Discussionmentioning

confidence: 99%

Cardiovascular magnetic resonance assessment of ventricular function and myocardial scarring before and early after repair of anomalous left coronary artery from the pulmonary artery

Latus

Gummel²,

Rupp³

et al. 2014

Journal of Cardiovascular Magnetic Resonance

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BackgroundIn patients with anomalous left coronary artery from the pulmonary artery (ALCAPA) left ventricular (LV) dilatation and dysfunction evolves due to diminished myocardial perfusion caused by coronary steal phenomenon. Using late gadolinium enhanced cardiovascular magnetic resonance (LGE-CMR) imaging, myocardial scarring has been shown in ALCAPA patients late after repair, however the incidence of scarring before surgery and its impact on postoperative course after surgical repair remained unknown.Methods8 ALCAPA-patients (mean age 10.0 ± 5.8 months) underwent CMR before and early after (mean 4.9 ± 2.5 months) coronary reimplantation procedures. CMR included functional analysis and LGE for detection of myocardial scars.ResultsLV dilatation (mean LVEDVI 171 ± 94 ml/m2) and dysfunction (mean LV-EF 22 ± 10 %) was present in all patients and improved significantly after surgery (mean LVEDV 68 ± 42 ml/m2, p = 0.02; mean LV-EF 58 ± 19 %, p < 0.001). Preoperative CMR revealed myocardial scarring in 2 of the 8 patients and did not predict postoperative course. At follow-up CMR, one LGE-positive patient showed delayed recovery of LV function while myocardial scarring was still present in both patients. In two patients new-onset transmural scarring was found, although functional recovery after operation was sufficient. One of them showed a stenosis of the left coronary artery and required resurgery.ConclusionsDespite diminished myocardial perfusion and severely compromised LV function, myocardial scarring was preoperatively only infrequently present. Improvement of myocardial function was independent of new-onset scarring while the impact of preoperative scarring still needs to be defined.

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Section: Discussionmentioning

confidence: 99%

Cardiovascular magnetic resonance assessment of ventricular function and myocardial scarring before and early after repair of anomalous left coronary artery from the pulmonary artery

Latus

Gummel²,

Rupp³

et al. 2014

Journal of Cardiovascular Magnetic Resonance

View full text Add to dashboard Cite

show abstract

“…Como alternativa, el diagnóstico puede certificarse mediante la visualización directa del origen de la ACI desde la AP mediante otras técnicas de imagen como la tomografía computarizada o la resonancia nuclear magnética. Esta última ha adquirido ventaja, ya que permite conocer la existencia de isquemia subendocárdica crónica, evaluar la función mitral y visualizar otros defectos congénitos asociados 12 . El tratamiento definitivo de esta anomalía es la cirugía.…”

Section: Discussionunclassified

Síndrome de ALCAPA en adulto: Caso clínico

et al. 2017

Rev. méd. Chile

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“…The presence of left ventricular dilatation, subendocardial scarring, and regional wall motion abnormalities are indicators of chronic ischemia and important substrate for malignant arrythmia. Evidence of delayed subendocardial enhancement on postgadolinium MRI imaging is thought to be an indication for surgical correction of the anomalous vessel …”

Section: Discussionmentioning

confidence: 99%

“…Cardiac MRI and electrocardiograph gated multislice computed tomographic angiography are reliable imaging modalities to use. Cardiac MRI has been increasingly utilized in multiple other studies to guide both diagnostic and therapeutic decisions in patients with ALCAPA . The presence of delayed subendocardial enhancement may be seen on cardiac MRI images.…”

Section: Discussionmentioning

confidence: 99%

“…Evidence of delayed subendocardial enhancement on postgadolinium MRI imaging is thought to be an indication for surgical correction of the anomalous vessel. 8,[13][14][15][16][17] The dysfunction of papillary muscles and adjacent myocardium seen on echocardiographic imaging provide information of mitral valve dysfunction. Cardiac MRI and electrocardiograph gated multislice computed tomographic angiography are reliable imaging modalities to use.…”

Section: Discussionmentioning

confidence: 99%

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The Management of the Older Adult Patient with Anomalous Left Coronary Artery from the Pulmonary Artery Syndrome: A Presentation of Two Cases and Review of the Literature

et al. 2013

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ALCAPA (anomalous left coronary artery from pulmonary artery) syndrome is a rare congenital abnormality that involves an anomalous insertion of the left coronary artery into the pulmonary artery. Ninety percent of patients present in the first year of life with signs and symptoms of heart failure or sudden cardiac death secondary to chronic myocardial ischemia. There have been an increasing number of reports of ALCAPA patients surviving to adulthood. There seems, however, to be a tendency to die suddenly in the third decade of life. Adult survivors are either asymptomatic or present with mitral regurgitation, cardiomyopathy, myocardial ischemia, or malignant arrhythmias. The management of the older patient presenting with symptoms resulting from ischemia and progressive left ventricular dysfunction remains a challenge. Treatment is largely based on guidelines for adult congenital heart disease management and an extrapolation of evidence from heart failure practice. Currently, surgical reimplantation of the anomalous coronary onto the aorta is the mainstay of treatment. The management of heart failure, sudden cardiac death, and ventricular arrhythmia present problems that are not addressed by reimplantation of the anomalous vessel alone. In this report, we present two cases with different modes of presentation and discuss treatment options.

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Magnetic resonance studies in management of adult cases with Bland-White-Garland syndrome

Cited by 16 publications

References 5 publications

Cardiovascular magnetic resonance assessment of ventricular function and myocardial scarring before and early after repair of anomalous left coronary artery from the pulmonary artery

Cardiovascular magnetic resonance assessment of ventricular function and myocardial scarring before and early after repair of anomalous left coronary artery from the pulmonary artery

Síndrome de ALCAPA en adulto: Caso clínico

The Management of the Older Adult Patient with Anomalous Left Coronary Artery from the Pulmonary Artery Syndrome: A Presentation of Two Cases and Review of the Literature

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