Hypoplastic coronary artery disease and hypertension in a child: a case report

Abstract

Background Hypoplastic coronary artery disease (HCAD) is an extremely rare disease associated with a risk of sudden cardiac death. It is rarely recognized in a live pediatric patient. Case summary We report a case of HCAD in a patient who first presented with vomiting and poor feeding, suggestive of acute heart failure due to cardiomyopathy or acute myocarditis in infancy. Hypertension and signs of ischemia became evident on electrocardiography and scintigraphy after his cardiac function fully recovered, and he was diagnosed with HCAD by angiography performed at the age of eight years. He has remained under close observation with anti-hypertensives, aspirin, and exercise restriction. Discussion Although HCAD is a rare disease, it may not only cause ischemia but may also result in heart failure and sudden cardiac death. It should be considered in any pediatric patient with heart failure. Mid-term follow-up visits might be necessary to detect signs of ischemia in pediatric patients presenting with features of heart failure.

Hypoplastic and Congenital Absence of Coronary Arteries and Its Correlation with Clinical Implications of Cardiac Circulation: A Systematic Review and Meta-Analysis

“…Eighty-eight studies were excluded due to discrepancies between their primary and secondary results compared to those of this review, or because they failed to meet the established criteria for good data extraction. Therefore, 39 articles [ 10 , 11 , 12 , 13 , 14 , 15 , 16 , 17 , 18 , 19 , 20 , 21 , 22 , 23 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 31 , 32 , 33 , 34 , 35 , 36 , 37 , 38 , 39 , 40 , 41 , 42 , 43 , 44 , 45 , 46 , 47 , 48 ] were selected for this study ( n = 140,137) (including patients, images, and cadavers) ( Figure 1 ).…”

Casting Light on The Hidden Prevalence: A Novel Perspective on Hypoplastic Coronary Artery Disease

“…Prior investigations failed to establish a concrete correlation between the presence of HCAD and the development of hypertension. Nevertheless, among 17 documented pediatric cases, two exhibited hypertension, posited as a compensatory mechanism at preserving coronary perfusion [28]. At the same time, it is important to remember that hypertension stands as a primary precipitant in the development of CAD.…”

Hypoplastic coronary artery disease, as a cause of sudden death

“…The significant reduction in the luminal diameter and length of one or more main coronary arteries characterizes hypoplastic coronary artery disease (HCAD). 14 , 15 According to the Indian population study on the normal diameters of the left main coronary artery, the proximal left anterior descending artery, proximal right coronary artery, and proximal left circumflex artery are respectively 4.08±0.44mm, 3.27±0.23mm, 3.20± 0.37mm, 2.97±0.37 mm. 16 Most of HCAD cases are more frequently detected during autopsies than by in vivo coronary angiography.…”

“…According to recent advanced studies on cases with HCAD, both congenital and acquired factors influence coronary artery flow. 14 , 15 The formation of the coronary arteries has been the subject of some research. It has been suggested that some genetic variables may be connected to HCAD.…”

“… 8 , 10 Implantable cardioverter-defibrillators are useful for aborting sudden cardiac death as secondary prevention. 7 , 14 , 15 , 24 Patients with HCAD who develop ischemic cardiomyopathy and end-stage cardiac failure may need a heart transplant. 21 Although HCAD is an entity with potential in vivo diagnosis, most cases are unveiled during autopsy.…”