Generation of Human-Induced Pluripotent Stem Cells from Wolfram Syndrome Type 2 Patients Bearing the c.103 + 1G>A CISD2 Mutation for Disease Modeling

Stem Cells Dev. 2018 Feb 15;27(4):287-295. doi: 10.1089/scd.2017.0158. Epub 2018 Jan 22.

Abstract

Wolfram syndrome (WFS) is a rare autosomal premature aging syndrome that shows signs of diabetes mellitus, optic atrophy, and deafness in addition to central nervous system and endocrine complications. The frequent form of WFS type 1 (WFS1) harbors causative mutations in the WFS1 gene, whereas the rare form or WFS type 2 (WFS2) involves CISD2. Mutations in these two genes are recognized by a subset of variable clinical symptoms and a set of overlapping features. In this study, we report on the generation of stable human-induced pluripotent stem cells (hiPSCs) derived from primary fibroblasts of a previously reported Italian family with CISD2 mutation (c.103 + 1G>A), occurring in the consensus intron 1 splicing site in two sisters, deleting the first exon of the transcript. The generated hiPSCs provide a cell model system to study the mutation's role in the multisystemic clinical disorders previously described and test eventual drug effects on the specific and associated clinical phenotype.

Keywords: Wolfram syndrome type 2; disease modeling; induced pluripotent stem cells.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Aging, Premature / genetics*
  • Aging, Premature / pathology
  • Cell Proliferation / genetics
  • Cells, Cultured
  • Family Health
  • Female
  • Fibroblasts / metabolism
  • Hearing Loss, Sensorineural / genetics*
  • Hearing Loss, Sensorineural / pathology
  • Humans
  • Induced Pluripotent Stem Cells / metabolism*
  • Introns / genetics
  • Male
  • Membrane Proteins / genetics*
  • Mitochondrial Diseases / genetics*
  • Mitochondrial Diseases / pathology
  • Mutation*
  • Optic Atrophy / genetics*
  • Optic Atrophy / pathology
  • RNA Splice Sites / genetics
  • Siblings

Substances

  • CISD2 protein, human
  • Membrane Proteins
  • RNA Splice Sites

Supplementary concepts

  • Wolfram Syndrome 2