Autosomal recessive spinocerebellar ataxia 20: Report of a new patient and review of literature

Eur J Med Genet. 2017 Feb;60(2):118-123. doi: 10.1016/j.ejmg.2016.11.006. Epub 2016 Nov 29.

Abstract

Inherited ataxias are an extremely heterogeneous group of disorders. Autosomal recessive spinocerebellar ataxia 20 (SCAR20) is a recently described disorder characterized by intellectual disability, ataxia, coarse facial features, progressive loss of Purkinje cells in the cerebellum and often hearing loss and skeletal abnormalities. Mutations in the gene SNX14, which plays an important role in autophagy, have been found to cause SCAR20. The unique clinical findings of progressive coarsening of facial features makes the clinical phenotype recognizable among the various hereditary ataxias. Here we report on a child with a novel missense mutation in the SNX14 gene that appears to be debilitating for protein conformation, function and review the previously reported cases from 15 families.

Keywords: Autophagy; Autosomal recessive spinocerebellar ataxia 20; Cerebellar atrophy; Hereditary ataxia; SNX14.

Publication types

  • Case Reports

MeSH terms

  • Autophagy / genetics
  • Cerebellum / physiopathology*
  • Child
  • Female
  • Humans
  • Mutation
  • Pedigree
  • Phenotype
  • Purkinje Cells / pathology*
  • Sorting Nexins / genetics*
  • Spinocerebellar Ataxias / genetics*
  • Spinocerebellar Ataxias / physiopathology

Substances

  • SNX14 protein, human
  • Sorting Nexins

Supplementary concepts

  • Spinocerebellar ataxia 20