Outcome of pulmonary and aortic stenosis in Williams-Beuren syndrome in an Asian cohort

Acta Paediatr. 2007 Jun;96(6):906-9. doi: 10.1111/j.1651-2227.2007.00308.x.

Abstract

Aims: To define the cardiovascular anomalies and the long-term outcomes in an Asian cohort with Williams-Beuren syndrome (WBS).

Methods: Data were retrieved from a retrospective chart review of patients who had a definitive diagnosis of WBS by fluorescence in situ hybridization between 1995 and 2005. All patients underwent echocardiography every 3-9 months. Ten patients underwent cardiac catheterization.

Results: Twenty-one patients with a total follow-up of 134 patient-years (median: 72 months) were enrolled. Characteristic dysmorphic facial features were noted in 19 patients (n = 19, 90%). All except one had associated cardiac anomalies, accounting for 0.3% (20/6640) of the patients with congenital heart disease. The spectrum of cardiac anomalies included supravalvular aortic stenosis (SVAS) (n = 15, 71%), peripheral pulmonary stenosis (PPS) (n = 12, 57%), pulmonary valve stenosis (PS) (n = 10, 47%), mitral valve prolapse (MVP) (n = 9, 43%), coarcation of the aorta (n = 4, 19%), ventricular septal defect (n = 2, 10%) and atrial septal defect (n = 1, 5%). Concurrent SVAS and PS/PPS were found in 14 (70%) patients. Only one patient required balloon dilation of PS, which improved. Regression of the stenoses occurred with a probability of 31, 90 and 71% at the age of 10 years for SAVS, PS and PPS, respectively.

Conclusions: Among our WBS patients, SVAS, PPS and PS were common, and were associated with probability of spontaneous regression, especially of right-sided lesions.

MeSH terms

  • Age Factors
  • Aortic Valve Stenosis / congenital*
  • Aortic Valve Stenosis / diagnostic imaging
  • Aortic Valve Stenosis / ethnology
  • Asian People*
  • Child
  • Child, Preschool
  • Disease Progression
  • Female
  • Humans
  • Infant
  • Male
  • Pulmonary Valve Stenosis / congenital*
  • Pulmonary Valve Stenosis / diagnostic imaging
  • Pulmonary Valve Stenosis / ethnology
  • Retrospective Studies
  • Ultrasonography
  • Williams Syndrome / complications
  • Williams Syndrome / ethnology*