Identification of a novel mutation in the APTX gene associated with ataxia-oculomotor apraxia

Cold Spring Harb Mol Case Stud. 2017 Nov 21;3(6):a002014. doi: 10.1101/mcs.a002014. Print 2017 Nov.

Abstract

Hereditary ataxias are a clinically and genetically heterogeneous family of disorders defined by the inability to control gait and muscle coordination. Given the nonspecific symptoms of many hereditary ataxias, precise diagnosis relies on molecular genetic testing. To this end, we conducted whole-exome sequencing (WES) on a large consanguineous Iranian family with hereditary ataxia and oculomotor apraxia. WES in five affected and six unaffected individuals resulted in the identification of a homozygous novel stop-gain mutation in the APTX gene (c.739A>T; p.Lys247*) that segregates with the phenotype. Mutations in the APTX (OMIM 606350) gene are associated with ataxia with oculomotor apraxia type 1 (OMIM 208920).

Keywords: apraxia; ataxia; athetosis; progressive cerebellar ataxia.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Apraxias / genetics*
  • Ataxia / complications
  • Ataxia / genetics
  • Cerebellar Ataxia / complications
  • Cerebellar Ataxia / congenital*
  • Cerebellar Ataxia / genetics
  • Child
  • Child, Preschool
  • Consanguinity
  • DNA-Binding Proteins / genetics*
  • DNA-Binding Proteins / metabolism
  • Female
  • Humans
  • Hypoalbuminemia / genetics*
  • Iran
  • Male
  • Middle Aged
  • Mutation / genetics
  • Nuclear Proteins / genetics*
  • Nuclear Proteins / metabolism
  • Pedigree
  • Phenotype
  • Spinocerebellar Degenerations / complications
  • Spinocerebellar Degenerations / genetics

Substances

  • APTX protein, human
  • DNA-Binding Proteins
  • Nuclear Proteins

Supplementary concepts

  • Early-onset ataxia with oculomotor apraxia and hypoalbuminemia