Phenotypic spectrum and sex effects in eleven myoclonus-dystonia families with epsilon-sarcoglycan mutations

Mov Disord. 2008 Mar 15;23(4):588-92. doi: 10.1002/mds.21785.

Abstract

Myoclonus-dystonia (M-D) due to SGCE mutations is characterized by early onset myoclonic jerks, often associated with dystonia. Penetrance is influenced by parental sex, but other sex effects have not been established. In 42 affected individuals from 11 families with identified mutations, we found that sex was highly associated with age at onset regardless of mutation type; the median age onset for girls was 5 years versus 8 years for boys (P < 0.0097). We found no association between mutation type and phenotype.

Publication types

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

MeSH terms

  • Adult
  • Aged
  • DNA Mutational Analysis
  • Dystonia / complications*
  • Dystonia / genetics*
  • Exons / genetics
  • Female
  • Humans
  • Introns / genetics
  • Male
  • Myoclonus / complications*
  • Myoclonus / genetics*
  • Phenotype*
  • Point Mutation / genetics
  • Sarcoglycans / genetics*
  • Severity of Illness Index
  • Sex Factors

Substances

  • SGCE protein, human
  • Sarcoglycans