Early-onset epileptic encephalopathy with myoclonic seizures related to 9q33.3-q34.11 deletion involving STXBP1 and SPTAN1 genes

Epileptic Disord. 2018 Jun 1;20(3):214-218. doi: 10.1684/epd.2018.0969.

Abstract

We describe a 10-month-old boy with early-onset epileptic encephalopathy who was found to have a hemizygous deletion in 9q33.3-q34.11 involving STXBP1 and SPTAN1 genes. He presented at the age of 2.5 months with frequent upper extremity myoclonus, hypotonia, and facial dysmorphisms. Interictal EEG showed multifocal polyspike and wave during wakefulness and sleep. Ictal EEG revealed low-amplitude generalized sharp slow activity, followed by diffuse attenuation. Metabolic testing was unrevealing. Brain MRI showed thinning of the corpus callosum with an absence of rostrum. This patient is the second reported case with 9q33.3-q34.11 deletion involving STXBP1 and SPTAN1 genes associated with epileptic encephalopathy and myoclonic seizures. Larger case series are needed to better delineate this association.

Keywords: Early Infantile Epileptic Encephalopathy; Early Myoclonic Encephalopathy; Ohtahara syndrome; SPTAN1; STXBP1; encephalopathy; microdeletion.

Publication types

  • Case Reports

MeSH terms

  • Brain / diagnostic imaging
  • Brain / physiopathology
  • Carrier Proteins / genetics*
  • Chromosome Deletion
  • Electroencephalography
  • Epilepsies, Myoclonic / diagnostic imaging
  • Epilepsies, Myoclonic / genetics*
  • Epilepsies, Myoclonic / physiopathology
  • Humans
  • Infant
  • Magnetic Resonance Imaging
  • Male
  • Microfilament Proteins / genetics*
  • Munc18 Proteins / genetics*
  • Spasms, Infantile / diagnostic imaging
  • Spasms, Infantile / genetics*
  • Spasms, Infantile / physiopathology

Substances

  • Carrier Proteins
  • Microfilament Proteins
  • Munc18 Proteins
  • STXBP1 protein, human
  • fodrin