Tdp-43 cryptic exons are highly variable between cell types

Mol Neurodegener. 2017 Feb 2;12(1):13. doi: 10.1186/s13024-016-0144-x.

Abstract

Background: TDP-43 proteinopathy is a prominent pathological feature that occurs in a number of human diseases including amyotrophic lateral sclerosis (ALS), frontotemporal dementia (FTD), and inclusion body myositis (IBM). Our recent finding that TDP-43 represses nonconserved cryptic exons led us to ask whether cell type-specific cryptic exons could exist to impact unique molecular pathways in brain or muscle.

Methods: In the present work, we investigated TDP-43's function in various mouse tissues to model disease pathogenesis. We generated mice to conditionally delete TDP-43 in excitatory neurons or skeletal myocytes and identified the cell type-specific cryptic exons associated with TDP-43 loss of function.

Results: Comparative analysis of nonconserved cryptic exons in various mouse cell types revealed that only some cryptic exons were common amongst stem cells, neurons, and myocytes; the majority of these nonconserved cryptic exons were cell type-specific.

Conclusions: Our results suggest that in human disease, TDP-43 loss of function may impair cell type-specific pathways.

Keywords: Amyotrophic lateral sclerosis; Bioinformatics; Frontotemporal dementia; Inclusion body myositis; TDP-43 –Nonconserved cryptic exons.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, U.S. Gov't, Non-P.H.S.
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • DNA-Binding Proteins / genetics*
  • Disease Models, Animal
  • Exons / genetics*
  • Immunoblotting
  • Immunohistochemistry
  • Mice
  • Mice, Knockout
  • Mice, Transgenic
  • Muscle Cells / metabolism*
  • Muscle Fibers, Skeletal / metabolism*
  • Neurons / metabolism*
  • Reverse Transcriptase Polymerase Chain Reaction
  • TDP-43 Proteinopathies / genetics

Substances

  • DNA-Binding Proteins
  • TDP-43 protein, mouse