Dynactin1 depletion leads to neuromuscular synapse instability and functional abnormalities.

ABSTRACT:

Background

Dynactin subunit 1 is the largest subunit of the dynactin complex, an activator of the molecular motor protein complex dynein. Reduced levels of DCTN1 mRNA and protein have been found in sporadic amyotrophic lateral sclerosis (ALS) patients, and mutations have been associated with disease, but the role of this protein in disease pathogenesis is still unknown.

Methods

We characterized a Dynactin1a depletion model in the zebrafish embryo and combined in vivo molecular analysis of primary motor neuron development with live in vivo axonal transport assays in single cells to investigate ALS-related defects. To probe neuromuscular junction (NMJ) function and organization we performed paired motor neuron-muscle electrophysiological recordings and GCaMP calcium imaging in

SUBMITTER: Bercier V 

PROVIDER: S-EPMC6617949 | biostudies-literature | 2019 Jul

REPOSITORIES: biostudies-literature