Heterogeneity of presentation and outcome in the Irish rapid-onset dystonia-parkinsonism kindred

Mov Disord. 2007 Jul 15;22(9):1325-7. doi: 10.1002/mds.21335.

Abstract

The authors report a 7-year follow-up video study and molecular data on the Irish rapid-onset dystonia-Parkinsonism kindred. All affected patients tested had a missense mutation in the Na(+)/K(+) -ATPase alpha3 subunit (ATP1A3), twice previously identified, suggestive of a mutation hotspot. Clinical presentation, progression, and outcome in this kindred is varied. Some patients remain stable over many years, others worsen, have a fluctuating course, or improve over time. To date there have been no effective treatments for this disorder, although Na(+)/K(+) ATPase may be a future therapeutic target. The broad phenotypic spectrum of RDP described in the text and detailed in the video, should be considered when evaluating patients with dystonia.

MeSH terms

  • Adolescent
  • Adult
  • Aged, 80 and over
  • Dystonia / complications*
  • Dystonia / genetics*
  • Family Health*
  • Female
  • Follow-Up Studies
  • Humans
  • Ireland
  • Male
  • Middle Aged
  • Mutation, Missense*
  • Parkinsonian Disorders / complications*
  • Parkinsonian Disorders / genetics*
  • Sodium-Potassium-Exchanging ATPase / genetics*

Substances

  • ATP1A3 protein, human
  • Sodium-Potassium-Exchanging ATPase