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Published on: October 4, 2014
Adult Movement Defects Associated with a CORL Mutation in Drosophila Display Behavioral Plasticity
Agapi Dimitriadou1, Nasia Chatzianastasi1, Panagiota I Zacharaki1
1Medical School, National and Kapodistrian University of Athens, 11527 Athens, Greece.
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 dCORL and dCORL 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.
Insights
Mutations in the CORL gene cause movement disorders in adult flies, but these defects improve with age. This age-dependent plasticity suggests new roles for CORL proteins in both flies and mammals.
Area of Science:
- Neuroscience
- Genetics
- Developmental Biology
Background:
- The CORL protein family, including Drosophila CORL, shares a Smad-binding region with mammalian SnoN and c-Ski.
- Human CORL proteins (Fussel15, Fussel18) have been linked to ataxia in genome-wide association studies.
- The precise roles of CORL proteins in mammals remain largely uncharacterized.
Purpose of the Study:
- To investigate the hypothesis that mutations in Drosophila CORL (dCORL) lead to adult movement disorders.
- To explore the age-dependent plasticity of movement defects caused by dCORL loss-of-function.
Main Methods:
- Comparative studies of adult Drosophila melanogaster with a dCORL deletion mutant (Df(4)dCORL) and control strains.
- Behavioral analyses including climbing ability and phototaxis.
- Generation of CRISPR-induced dCORL mutations to confirm observed phenotypes.
- Assessment of courtship behavior in grouped versus single-reared males.
Main Results:
- Loss of dCORL causes significant climbing and phototaxis defects in adult flies.
- These movement defects exhibit age-dependent plasticity, with climbing defects being eliminated and phototaxis defects partially ameliorated by age.
- CRISPR-generated mutations confirmed the climbing and phototaxis defects associated with dCORL.
- Group rearing in males led to reduced courtship index, but this was independent of dCORL function.
Conclusions:
- Loss of dCORL function in Drosophila results in age-dependent plasticity of adult movement disorders.
- These findings suggest novel hypotheses regarding the functions of CORL proteins in both invertebrates and vertebrates.
- The study highlights the complex interplay between genetics and aging in neurological function.
