A novel inhibitor rescues cerebellar defects in a zebrafish model of Down syndrome-associated kinase Dyrk1A overexpression.

ABSTRACT: The highly conserved dual-specificity tyrosine phosphorylation-regulated kinase 1A (Dyrk1A) plays crucial roles during central nervous system development and homeostasis. Furthermore, its hyperactivity is considered responsible for some neurological defects in individuals with Down syndrome. We set out to establish a zebrafish model expressing human Dyrk1A that could be further used to characterize the interaction between Dyrk1A and neurological phenotypes. First, we revealed the prominent expression of dyrk1a homologs in cerebellar neurons in the zebrafish larval and adult brains. Overexpression of human dyrk1a in postmitotic cerebellar Purkinje neurons resulted in a structural misorganization of the Purkinje cells in cerebellar hemispheres and a compaction of this cell population. This i

SUBMITTER: Buchberger A 

PROVIDER: S-EPMC8239740 | biostudies-literature | 2021 Jul

REPOSITORIES: biostudies-literature