Analysis of ATP13A2 in large neurodegeneration with brain iron accumulation (NBIA) and dystonia-parkinsonism cohorts

Neurosci Lett. 2012 Aug 8;523(1):35-8. doi: 10.1016/j.neulet.2012.06.036. Epub 2012 Jun 25.

Abstract

Several causative genes have been identified for both dystonia-parkinsonism and neurodegeneration with brain iron accumulation (NBIA), yet many patients do not have mutations in any of the known genes. Mutations in the ATP13A2 lead to Kufor Rakeb disease, a form of autosomal recessive juvenile parkinsonism that also features oromandibular dystonia. More recently, evidence of iron deposition in the caudate and putamen have been reported in patients with ATP13A2 mutations. We set out to determine the frequency of ATP13A2 mutations in cohorts of idiopathic NBIA and dystonia-parkinsonism. We screened for large deletions using whole genome arrays, and sequenced the entire coding region in 92 cases of NBIA and 76 cases of dystonia-parkinsonism. A number of coding and non-coding sequence variants were identified in a heterozygous state, but none were predicted to be pathogenic based on in silico analyses. Our results indicate that ATP13A2 mutations are a rare cause of both NBIA and dystonia-parkinsonism.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Aged
  • Aged, 80 and over
  • Cohort Studies
  • Comorbidity
  • Dystonic Disorders / epidemiology*
  • Dystonic Disorders / genetics*
  • Female
  • Genetic Markers / genetics
  • Genetic Predisposition to Disease / epidemiology*
  • Genetic Predisposition to Disease / genetics*
  • Group VI Phospholipases A2 / deficiency
  • Group VI Phospholipases A2 / genetics
  • Humans
  • Internationality
  • Iron Metabolism Disorders / epidemiology*
  • Iron Metabolism Disorders / genetics*
  • Male
  • Middle Aged
  • Mutation / genetics
  • Neuroaxonal Dystrophies / epidemiology*
  • Neuroaxonal Dystrophies / genetics*
  • Parkinsonian Disorders / epidemiology*
  • Parkinsonian Disorders / genetics*
  • Polymorphism, Single Nucleotide / genetics
  • Prevalence
  • Proton-Translocating ATPases / genetics*
  • Risk Factors
  • Young Adult

Substances

  • ATP13A2 protein, human
  • Genetic Markers
  • Group VI Phospholipases A2
  • Proton-Translocating ATPases

Supplementary concepts

  • Dystonia-Parkinsonism, Adult-Onset
  • Neurodegeneration with brain iron accumulation (NBIA)