First report of atretic coronary sinus stenting in a 5-kg infant resulting in dramatic improvement of ventricular function in functional single ventricle

Congenit Heart Dis. 2014 Nov-Dec;9(6):E175-8. doi: 10.1111/chd.12118. Epub 2013 Jul 3.

Abstract

The patient was a male infant with L-transposition of great arteries (L-TGA), Ebstein's anomaly of the tricuspid valve, subvalvar aortic stenosis, ventricular septal defect (VSD), hypoplastic right ventricle, arch hypoplasia, and congenital complete heart block. He underwent a Norwood procedure, aortic arch repair, permanent pacemaker implantation, and a 3.5-mm aortopulmonary shunt at 4 days of age. At the time of his surgery, left ventricular function was in the normal range (ejection fraction [EF] = 67%). However by 3 months of age, he was noted to have developed moderate-severe biventricular dysfunction (left ventricular ejection fraction [LVEF] = 34%). Atresia of the coronary sinus with a small left superior venacava (LSVC) and a bridging vein was discovered during cardiac catheterization at this time. The coronary sinus mean pressure was 17 mm Hg, and the common atrial mean pressure was 6 mmHg. We opened the atretic coronary sinus ostium using radiofrequency ablation and stent placement. There was dramatic improvement in ventricular function observed over a 2-month period. Follow-up cardiac catheterization 5 months later revealed the stent in the coronary sinus to be widely patent with no intimal buildup, and the ventricular function was normal (LVEF = 58%). The patient had a bidirectional Glenn procedure with an uncomplicated postoperative course and is currently awaiting Fontan completion.

Keywords: Atretic Coronary Sinus; Functional Single Ventricle; Stent Implantation.

Publication types

  • Case Reports

MeSH terms

  • Abnormalities, Multiple*
  • Angioplasty, Balloon, Coronary / instrumentation*
  • Body Weight
  • Cineangiography
  • Coronary Angiography
  • Coronary Sinus / abnormalities*
  • Coronary Sinus / physiopathology
  • Coronary Vessel Anomalies / complications
  • Coronary Vessel Anomalies / diagnosis
  • Coronary Vessel Anomalies / physiopathology
  • Coronary Vessel Anomalies / therapy*
  • Heart Defects, Congenital / complications
  • Heart Defects, Congenital / diagnosis
  • Heart Defects, Congenital / physiopathology*
  • Heart Ventricles / abnormalities*
  • Heart Ventricles / physiopathology
  • Humans
  • Infant
  • Infant, Newborn
  • Male
  • Radiography, Interventional / methods
  • Recovery of Function
  • Stents*
  • Stroke Volume
  • Time Factors
  • Treatment Outcome
  • Ventricular Function, Left*
  • Ventricular Function, Right*

Supplementary concepts

  • Right ventricle hypoplasia