TY - JOUR
T1 - Adult movement defects associated with a CORL mutation in Drosophila display behavioral plasticity
AU - Dimitriadou, Agapi
AU - Chatzianastasi, Nasia
AU - Zacharaki, Panagiota I.
AU - O'Connor, Mary Jane
AU - Goldsmith, Samuel L.
AU - O'Connor, Michael B.
AU - Consoulas, Christos
AU - Newfeld, Stuart J.
N1 - Publisher Copyright:
© The Author(s) 2020. Published by the Genetics Society of America.
PY - 2020/4/1
Y1 - 2020/4/1
N2 - 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.
AB - 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.
KW - Ataxia
KW - Climbing
KW - Courtship
KW - Fussel/SKOR
KW - Phototaxis
UR - http://www.scopus.com/inward/record.url?scp=85084271989&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=85084271989&partnerID=8YFLogxK
U2 - 10.1534/g3.120.400648
DO - 10.1534/g3.120.400648
M3 - Article
C2 - 32161085
AN - SCOPUS:85084271989
SN - 2160-1836
VL - 10
SP - 1697
EP - 1706
JO - G3: Genes, Genomes, Genetics
JF - G3: Genes, Genomes, Genetics
IS - 4
ER -