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Review
. 2017 Jan 31;11(1):24.
doi: 10.1186/s13256-016-1161-7.

Hypertrophic obstructive cardiomyopathy with multiple coronary arteries to right ventricular microfistulas: a case report and review of the literature

Affiliations
Review

Hypertrophic obstructive cardiomyopathy with multiple coronary arteries to right ventricular microfistulas: a case report and review of the literature

Daulat Singh Meena et al. J Med Case Rep. .

Abstract

Background: Coronary artery microfistulas are a rare anomaly; their association with hypertrophic cardiomyopathy is even rarer and can lead to serious cardiac complications owing to coronary steal phenomena such as angina pectoris, myocardial infarction, congestive heart failure, ventricular and supraventricular arrhythmias, syncope, and sudden death.

Case presentation: A 32-year-old Indian woman presented to our institute with severe angina on exertion and multiple episodes of pre-syncope. Echocardiography revealed hypertrophic obstructive cardiomyopathy. Coronary angiography showed no significant atherosclerotic lesions; however, it revealed multiple microfistulas originated from all three major coronary arteries and draining into her right ventricle. This finding was confirmed by the rapid filling of the pulmonary artery after dye was injected into her left coronary artery during a cardiac catheterization study and by a significant oxygen step up of 15 % seen from her right atria to right ventricle during oximetry analysis. We treated our patient's condition with medical therapy including metoprolol and nicorandil. She improved and angina grade had decreased from class III to class II on a follow-up visit 1 month after discharge.

Conclusions: In this case report and literature review, we highlight an unusual but important association that can lead to symptomatic worsening of angina in young patients with hypertrophic cardiomyopathy owing to coronary steal phenomena.

Keywords: Angina in young; Congenital coronary anomaly; Coronary angiography; HOCM.

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Figures

Fig. 1
Fig. 1
a Echocardiography revealed asymmetrical hypertrophic obstructive cardiomyopathy. b Pulsed wave Doppler suggested a resting sub-valvular obstruction with a pressure gradient of 33 mmHg
Fig. 2
Fig. 2
Multiple coronary artery microfistulas arising from the left anterior descending coronary artery and left circumflex artery
Fig. 3
Fig. 3
a Multiple coronary artery microfistulas arising from the right coronary artery to the right ventricle. b Left coronary artery microfistula draining to the right ventricle as shown by the filling of the artery (black arrow) from the left anterior descending coronary artery and left circumflex artery

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