Novel Homozygous Deletion in STRADA Gene Associated With Polyhydramnios, Megalencephaly, and Epilepsy in 2 Siblings: Implications for Diagnosis and Treatment

J Child Neurol. 2018 Dec;33(14):925-929. doi: 10.1177/0883073818802724. Epub 2018 Oct 12.

Abstract

Mutations in the STE20-related kinase adaptor α ( STRADA) gene have been reported to cause an autosomal recessive neurodevelopmental disorder characterized by infantile-onset epilepsy, developmental delay, and craniofacial dysmorphisms. To date, there have been 17 reported individuals diagnosed with STRADA mutations, 16 of which are from a single Old Order Mennonite cohort and share a deletion of exons 9-13. The remaining individual is of consanguineous Indian descent and has a homozygous single-base pair duplication. We report a novel STRADA gene deletion of exons 7-9 in 2 sisters from nonconsanguineous parents, as well as an improvement in seizure control in 1 sibling following treatment with sirolimus, an m-Tor inhibitor of potential benefit to patients with this genetic mutation.

Keywords: autosomal recessive; epileptic encephalopathy; facial dysmorphism; neonatal seizures; nephrocalcinosis; neurodevelopment; next-generation sequencing; seizures.

MeSH terms

  • Adaptor Proteins, Signal Transducing / genetics*
  • Adolescent
  • Electroencephalography
  • Epilepsy / complications*
  • Epilepsy / diagnostic imaging
  • Epilepsy / genetics*
  • Female
  • Humans
  • Magnetic Resonance Imaging
  • Megalencephaly / complications*
  • Megalencephaly / diagnostic imaging
  • Megalencephaly / genetics*
  • Polyhydramnios / diagnostic imaging
  • Polyhydramnios / genetics*
  • Pregnancy
  • Sequence Deletion / genetics*
  • Siblings

Substances

  • Adaptor Proteins, Signal Transducing