Adult Movement Defects Associated with a CORL Mutation in Drosophila Display Behavioral Plasticity

Abstract

The CORL family of CNS-specific proteins share a Smad-binding region with mammalian SnoN and c-Ski protooncogenes. In this family Drosophila CORL has two mouse and two human relatives. Roles for the mouse and human CORL proteins are largely unknown. Based on genome-wide association studies linking the human CORL proteins Fussel15 and Fussel18 with ataxia, we tested the hypothesis that dCORL mutations will cause adult movement disorders. For our initial tests, we conducted side by side studies of adults with the small deletion Df(4)dCORL and eight control strains. We found that deletion mutants exhibit three types of behavioral plasticity. First, significant climbing defects attributable to loss of dCORL are eliminated by age. Second, significant phototaxis defects due to loss of dCORL are partially ameliorated by age and are not due to faulty photoreceptors. Third, Df(4)dCORL males raised in groups have a lower courtship index than males raised as singles though this defect is not due to loss of dCORL. Subsequent tests showed that the climbing and phototaxis defects were phenocpied by dCORL21B and dCORL23C two CRISPR generated mutations. Overall, the finding that adult movement defects due to loss of dCORL are subject to age-dependent plasticity suggests new hypotheses for CORL functions in flies and mammals.

New resources for the Drosophila 4th chromosome: FRT101F enabled mitotic clones and Bloom syndrome helicase enabled meiotic recombination

“…Among the human homologs are conserved signaling molecules in the Hedgehog ( cubitus interruptus and fd102C are GLI and FOXL1), Wingless ( pangolin and legless are TCF and BCL9), and TGF-β pathways ( myoglianin and activin-β are Myostatin and INHBB). There are also numerous neuronal genes: unc-13 (UNC13A regulates α-secretase; Rossner et al 2004 ), sox102f (SOX5 causes Lamb-Shaffer syndrome; Lamb et al 2012 ), mGluR (GRM3 mutations may cause memory loss; de Quervain and Papassotiropoulos 2006 ) and dCORL ( fussel in Flybase; SKOR1 is linked to ataxia; Dimitriadou et al 2020 ).…”