Novel SPTBN2 gene mutation and first intragenic deletion in early onset spinocerebellar ataxia type 5

Ann Clin Transl Neurol. 2021 Apr;8(4):956-963. doi: 10.1002/acn3.51345. Epub 2021 Mar 23.

Abstract

In the present study, we describe two novel cases of SCA5 with early onset. The first one, carrying a novel heterozygous de novo missense mutation in SPTBN2 gene, showed a striking very severe cerebellar atrophy and reduction of volume of the pons at a very young age (16 months). The latter, carrying the first de novo intragenic deletion so far reported in SPTBN2 gene, showed a mild cerebellar atrophy involving the hemispheres and a later onset. In both cases, for the first time, a hyperintense signal of the dentate nuclei was observed.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Age of Onset
  • Child, Preschool
  • Female
  • Gene Deletion
  • Humans
  • Infant
  • Magnetic Resonance Imaging
  • Male
  • Pedigree
  • Spectrin / genetics*
  • Spinocerebellar Ataxias / genetics*
  • Spinocerebellar Ataxias / pathology*

Substances

  • SPTBN2 protein, human
  • Spectrin

Grants and funding

This work was funded by Italian Ministry of Health grants RC2020, RC021, RF‐NET‐2013‐02356160, and 5X MILLE; Fondazione Regionale Lombarda per la Ricerca Biomedica grant Care4Neurorare.