Evaluation of parkinsonism and striatal dopamine transporter loss in patients with spinocerebellar ataxia type 6

J Neurol. 2016 Nov;263(11):2302-2307. doi: 10.1007/s00415-016-8261-6. Epub 2016 Aug 20.

Abstract

It is unclear whether patients with spinocerebellar ataxia type 6 (SCA6) have parkinsonism and striatal dopamine transporter (DAT) loss, based on previously small size studies without well-matched controls. A study with a larger number of patients and both age- and gender-matched healthy controls (HCs) is needed for a better answer to this question. Twelve genetically confirmed ataxic SCA6 patients (six male six female, age 65.3 ± 11.2 years), and eight age- and gender-matched HCs (five male three female, age 71.3 ± 8.6 years) were enrolled during 2013-2015 from tertiary movement disorders and ataxia clinics. Clinical assessment for parkinsonism, and qualitative and quantitative assessment of DAT level on DaTscan™ imaging were conducted in SCA6 patients compared to HCs. We found no convincing parkinsonism in SCA6 patients, given generalized bradykinesia in the context of significant ataxia in all, with mild symmetric rigidity in five without resting tremor. Furthermore, we found no striatal DAT loss in anterior, posterior, and total putamen and caudate on imaging, assessed independently by qualitative visual inspection in a blinded manner by the nuclear medicine specialist and movement disorder specialist (kappa = 1). Additional quantitative analysis on these areas did not reveal significant DAT loss either in SCA6 patients compared to HCs. We conclude that there is no convincing parkinsonism or DAT loss in SCA6 patients in this unique study with a larger than previously reported number of patients compared to both age- and gender-matched HCs, suggesting that dopaminergic dysfunction is not usually involved in SCA6.

Keywords: Dopamine transporter; Parkinsonism; SCA6; Spinocerebellar ataxia.

MeSH terms

  • Aged
  • Aged, 80 and over
  • Calcium Channels / genetics
  • Case-Control Studies
  • Corpus Striatum
  • Dopamine Plasma Membrane Transport Proteins / metabolism*
  • Female
  • Humans
  • Image Processing, Computer-Assisted
  • Male
  • Middle Aged
  • Parkinsonian Disorders / complications*
  • Positron-Emission Tomography
  • Spinocerebellar Ataxias / complications*
  • Spinocerebellar Ataxias / genetics
  • Trinucleotide Repeats / genetics

Substances

  • CACNA1A protein, human
  • Calcium Channels
  • Dopamine Plasma Membrane Transport Proteins