Sudden cardiac arrest during anesthesia in a 30-month-old boy with syndactyly: a case of genetically proven Timothy syndrome

J Korean Med Sci. 2013 May;28(5):788-91. doi: 10.3346/jkms.2013.28.5.788. Epub 2013 May 2.

Abstract

Timothy syndrome, long QT syndrome type 8, is highly malignant with ventricular tachyarrhythmia. A 30-month-old boy had sudden cardiac arrest during anesthesia induction before plastic surgery for bilateral cutaneous syndactyly. After successful resuscitation, prolonged QT interval (QTc, 0.58-0.60 sec) and T-wave alternans were found in his electrocardiogram. Starting β-blocker to prevent further tachycardia and collapse event, then there were no more arrhythmic events. The genes KCNQ1, KCNH2, KCNE1 and 2, and SCN5A were negative for long QT syndrome. The mutation p.Gly406Arg was confirmed in CACNA1C, which maintains L-type calcium channel depolarization in the heart and other systems.

Keywords: Long QT Syndrome; Syndactyly.

Publication types

  • Case Reports

MeSH terms

  • Anesthesia / adverse effects*
  • Autistic Disorder
  • Calcium Channels, L-Type / genetics*
  • Death, Sudden, Cardiac / etiology*
  • Electroencephalography
  • Humans
  • Infant
  • Long QT Syndrome / genetics*
  • Magnetic Resonance Imaging
  • Male
  • Methyl Ethers / adverse effects
  • Nitric Oxide / adverse effects
  • Polymorphism, Single Nucleotide
  • Sequence Analysis, DNA
  • Sevoflurane
  • Surgery, Plastic
  • Syndactyly / diagnosis
  • Syndactyly / genetics*
  • Syndactyly / surgery

Substances

  • CACNA1C protein, human
  • Calcium Channels, L-Type
  • Methyl Ethers
  • Nitric Oxide
  • Sevoflurane

Supplementary concepts

  • Timothy syndrome