Severe episodic neurological deficits and permanent mental retardation in a child with a novel FHM2 ATP1A2 mutation

Ann Neurol. 2006 Feb;59(2):310-4. doi: 10.1002/ana.20760.

Abstract

Objective: Attacks of familial hemiplegic migraine (FHM) are usually associated with transient, completely reversible symptoms. Here, we studied the ATP1A2 FHM2 gene in a young girl with episodes of both very severe and transient neurological symptoms that were triggered by mild head trauma as well as permanent mental retardation. Her family members suffered from hemiplegic and confusional migraine attacks.

Methods: Mutation analysis of the ATP1A2 gene was performed by direct sequencing of all exons and flanking intronic regions, using genomic DNA of the proband. Functional consequences of the mutation were analyzed by cellular survival assays.

Results: We identified a novel G615R ATP1A2 mutation in the proband and several of her family members. Functional analysis of mutant Na,K-ATPase in cellular survival assays showed a complete loss-of-function effect.

Interpretation: Permanent mental retardation in children may be caused by ATP1A2 mutations.

Publication types

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

MeSH terms

  • Arginine / genetics
  • Blotting, Northern / methods
  • Blotting, Western / methods
  • Child
  • DNA Mutational Analysis / methods
  • Electroencephalography / methods
  • Female
  • Gene Expression / physiology
  • Glycine / genetics
  • HeLa Cells
  • Humans
  • Intellectual Disability / genetics*
  • Intellectual Disability / pathology
  • Magnetic Resonance Imaging / methods
  • Migraine with Aura / genetics*
  • Migraine with Aura / pathology
  • Migraine with Aura / physiopathology
  • Mutagenesis / physiology
  • Mutation*
  • Sodium-Potassium-Exchanging ATPase / genetics*
  • Transfection / methods

Substances

  • Arginine
  • ATP1A2 protein, human
  • Sodium-Potassium-Exchanging ATPase
  • Glycine