Intestinal dysmotility in a zebrafish (Danio rerio) shank3a;shank3b mutant model of autism.

ABSTRACT:

Background and aims

Autism spectrum disorder (ASD) is currently estimated to affect more than 1% of the world population. For people with ASD, gastrointestinal (GI) distress is a commonly reported but a poorly understood co-occurring symptom. Here, we investigate the physiological basis for GI distress in ASD by studying gut function in a zebrafish model of Phelan-McDermid syndrome (PMS), a condition caused by mutations in the SHANK3 gene.

Methods

To generate a zebrafish model of PMS, we used CRISPR/Cas9 to introduce clinically related C-terminal frameshift mutations in shank3a and shank3b zebrafish paralogues (shank3abΔC). Because PMS is caused by SHANK3 haploinsufficiency, we assessed the digestive tract (DT) structure and function in zeb

SUBMITTER: James DM 

PROVIDER: S-EPMC6357389 | biostudies-literature | 2019

REPOSITORIES: biostudies-literature