CAG repeat expansions in patients with sporadic cerebellar ataxia

Acta Neurol Scand. 1998 Jul;98(1):55-9. doi: 10.1111/j.1600-0404.1998.tb07378.x.

Abstract

CAG repeat expansions cause spinocerebellar ataxia type 1 (SCA1), SCA2, SCA3, SCA6 and dentatorubral-pallidoluysian atrophy (DRPLA). So far these expansions have been examined mainly in ataxia patients with a family history. However, some sporadic cases with SCA have recently been reported. To elucidate the frequency and characteristics of sporadic SCAs, we screened 85 Japanese ataxia patients without a family history for the SCA1, SCA2, SCA3, SCA6 and DRPLA mutations. As a result, 19 patients (22%) were found to have expanded CAG repeats. Among sporadic SCAs, the SCA6 mutation was most frequently observed. The sporadic SCA6 patients had smaller CAG repeats and a later age of onset than SCA6 patients with an established family history. We also identified one father-child pair in which intermediate sized CAG repeats expanded into the SCA2 disease range during transmission. These findings suggest that patients with ataxia even without a family history should be examined for a CAG repeat expansion.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Age Factors
  • Aged
  • Aged, 80 and over
  • DNA Mutational Analysis
  • Female
  • Gene Frequency / genetics
  • Genetic Carrier Screening
  • Humans
  • Japan
  • Male
  • Middle Aged
  • Polymerase Chain Reaction
  • Spinocerebellar Degenerations / diagnosis
  • Spinocerebellar Degenerations / genetics*
  • Trinucleotide Repeats / genetics*