Hypoplastic left heart syndrome with regressed ventriculocoronary fistulae after the Norwood operation
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CASE REPORT
Hypoplastic left heart syndrome with regressed ventriculocoronary fistulae after the Norwood operation S. Matsuo1 · N. Masaki1 · M. Mizumoto1 · S. Sai1 Received: 8 August 2019 / Accepted: 22 November 2019 © The Japanese Association for Thoracic Surgery 2019
Abstract Left ventriculocoronary artery fistulae can cause deterioration of postoperative outcomes in patients with hypoplastic left heart syndrome. We successfully performed the Norwood operation with right ventricle-pulmonary artery shunt without a cardiac arrest in an infant with hypoplastic left heart syndrome and large coronary artery fistulae. Temporary postoperative right ventricular dysfunction gradually improved, and left ventricular volume decreased by the time of bidirectional Glenn shunt procedure. Left ventriculocoronary artery fistulae regressed after the Norwood operation, illustrating that large coronary artery fistulae can regress over time following right ventricular decompression. Keywords Left ventriculocoronary artery fistulae · Norwood operation · Bidirectional Glenn shunt procedure · Right ventricular decompression
Introduction Mortality rates are higher in patients with the mitral stenosis–aortic atresia (MS–AA) variant of hypoplastic left heart syndrome (HLHS) than those with other variants [1, 2]. The relationship between high mortality rates and coronary abnormalities, such as left ventriculocoronary artery fistulae (LVCAF), which are prevalent in HLHS patients with MS–AA, is concerning because it may cause coronary malperfusion and myocardial ischemia, leading to early deterioration, especially during cardiopulmonary bypass [2, 3]. Herein, we present the case of an infant with HLHS whose LVCAF regressed following the modified Norwood operation that was completed without a cardiac arrest (CA).
Case A full-term male newborn weighing 2956 g with a prenatal diagnosis of HLHS was transferred to our hospital on day 1 after birth. The patient was diagnosed with the MS–AA variant of HLHS. Further, echocardiography revealed the * S. Matsuo [email protected] 1
Department of Cardiovascular Surgery, Miyagi Children’s Hospital, 4‑3‑17 Ochiai, Aoba‑ku, Sendai 989‑3126, Japan
LVCAF (Fig. 1a). The retrograde blood flow from LVCAF in systole at the ventricular septal wall was observed. The size of the atrial septal defect was 2.5 mm. Cardiac catheterization on day 2 showed a retrograde flow of the right coronary artery through the fistula from the left ventricle (LV) by left ventriculography (Fig. 2a). Cardiac catheterization showed a retrograde flow of the right coronary artery through the fistula from the left ventricle (LV) by left ventriculography and aortography. And antegrade flow in diastole and retrograde flow in systole were observed. The left atrial pressure was 8 mmHg. Interatrial pressure gradient was 5 mmHg. LV pressure was over-systemic. The ratio of left and right ventricular (LV/RV) pressures was 2.2. Balloon atrial septostomy (BAS) was not effective for restrictive atrial septal defect (ASD). Bilatera
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