Seizures as presenting and prominent symptom in chorea-acanthocytosis with c.2343del VPS13A gene mutation

Epilepsia. 2016 Apr;57(4):549-56. doi: 10.1111/epi.13318. Epub 2016 Jan 27.

Abstract

Objective: The aim of the study was to characterize the clinical features of nine patients in three families with chorea-acanthocytosis (ChAc) sharing the same rare c.2343del mutation in the VPS13A gene.

Methods: Genetic test results, clinical description, magnetic resonance imaging (MRI), and electroencephalography (EEG), as well as laboratory results are summarized.

Results: ChAc is a rare genetic disorder characterized by hyperkinetic movements, seizures, cognitive decline, neuropsychiatric symptoms, and acanthocytes on peripheral blood smear. This unique cohort of nine patients is characterized by seizures as a first and prominent symptom. In our patients, other features of ChAc appeared later, including tics, other movement disorders, dysarthria, and mild to moderate cognitive decline.

Significance: Patients with chorea-acanthocytosis carrying the described rare mutation can present with focal, treatment-resistant seizures.

Keywords: Chorea-acanthocytosis; Chorein; Epilepsy; Genetics; VPS13A.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Diagnosis, Differential
  • Electroencephalography / methods
  • Female
  • Humans
  • Magnetic Resonance Imaging / methods
  • Male
  • Mutation / genetics*
  • Neuroacanthocytosis / complications
  • Neuroacanthocytosis / diagnosis*
  • Neuroacanthocytosis / genetics*
  • Pedigree
  • Seizures / diagnosis*
  • Seizures / etiology
  • Seizures / genetics*
  • Vesicular Transport Proteins / genetics*
  • Young Adult

Substances

  • VPS13A protein, human
  • Vesicular Transport Proteins

Associated data

  • GENBANK/NC_000009.11
  • GENBANK/NM_015186.3
  • GENBANK/NM_033305.2