Blepharospasm and limb dystonia caused by Mohr-Tranebjaerg syndrome with a novel splice-site mutation in the deafness/dystonia peptide gene

Mov Disord. 2007 Jul 15;22(9):1328-31. doi: 10.1002/mds.21351.

Abstract

Mohr-Tranebjaerg syndrome (MTS) is an X-linked disorder characterized by childhood-onset progressive deafness, dystonia, spasticity, mental deterioration, and blindness. It is due to mutations in the deafness/dystonia peptide (DDP1) gene. We describe a sporadic 42-year-old man with MTS presenting with postlingual deafness, adult-onset progressive dystonia with marked arm tremor, mild spasticity of the legs, and visual disturbance due to a novel mutation (g to a transition at the invariant gt of the 5' splice donor site of exon 1) in the DDP1 gene. This case, and a review of previously reported cases, highlights a variety of potential diagnostic pitfalls in this condition.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Blepharospasm / etiology
  • Blepharospasm / genetics*
  • DNA Mutational Analysis / methods
  • Dystonia / etiology
  • Dystonia / genetics*
  • Humans
  • Male
  • Membrane Transport Proteins / genetics*
  • Mitochondrial Precursor Protein Import Complex Proteins
  • Mutation / genetics*
  • Orofaciodigital Syndromes / complications
  • Orofaciodigital Syndromes / genetics*
  • RNA Splice Sites / genetics*

Substances

  • Membrane Transport Proteins
  • Mitochondrial Precursor Protein Import Complex Proteins
  • RNA Splice Sites
  • TIMM8A protein, human