Novel SIL1 mutations cause cerebellar ataxia and atrophy in a French-Canadian family

Neurogenetics. 2015 Oct;16(4):315-8. doi: 10.1007/s10048-015-0455-z. Epub 2015 Aug 11.

Abstract

Two French-Canadian sibs with cerebellar ataxia and dysarthria were seen in our neurogenetics clinic. The older brother had global developmental delay and spastic paraplegia. Brain MRIs from these two affected individuals showed moderate to severe cerebellar atrophy. To identify the genetic basis for their disease, we conducted a whole exome sequencing (WES) investigation using genomic DNA prepared from the affected sibs and their healthy father. We identified two mutations in the SIL1 gene, which is reported to cause Marinesco-Sjögren syndrome. This study emphasizes how the diagnosis of patients with ataxic gait and cerebellar atrophy may benefit from WES to identify the genetic cause of their condition.

Keywords: Ataxia; Cerebellar; Mutations; SIL1; Whole exome sequencing.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Atrophy
  • Canada
  • Cerebellar Ataxia / complications
  • Cerebellar Ataxia / genetics*
  • Cerebellar Ataxia / pathology*
  • Cerebellum / pathology
  • Dysarthria / complications
  • Dysarthria / genetics*
  • Dysarthria / pathology*
  • Female
  • Genes, Recessive
  • Guanine Nucleotide Exchange Factors / genetics*
  • Humans
  • Male
  • Mutation*
  • Siblings
  • Young Adult

Substances

  • Guanine Nucleotide Exchange Factors
  • SIL1 protein, human