Drosophila lacking dfmr1 activity show defects in circadian output and fail to maintain courtship interest

Thomas C. Dockendorff; Henry S. Su; Sean M.J. McBride; Zhaohai Yang; Catherine H. Choi; Kathleen K. Siwicki; Amita Sehgal; Thomas A. Jongens

doi:10.1016/S0896-6273(02)00724-9

Drosophila lacking dfmr1 activity show defects in circadian output and fail to maintain courtship interest

Thomas C. Dockendorff, Henry S. Su, Sean M.J. McBride, Zhaohai Yang, Catherine H. Choi, Kathleen K. Siwicki, Amita Sehgal, Thomas A. Jongens

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233 Scopus citations

Abstract

Fragile X mental retardation is a prominent genetic disorder caused by the lack of the FMR1 gene product a known RNA binding protein. Specific physiologic pathways regulated by FMR1 function have yet to be identified. Adult dfmr1 (also called dfxr) mutant flies display arrhythmic circadian activity and have erratic patterns of locomotor activity whereas overexpression of dFMR1 leads to a lengthened period. dfmr1 mutant males also display reduced courtship activity which appears to result from their inability to maintain courtship interest. Molecular analysis fails to reveal any defects in the expression of clock components; however the CREB output is affected. Morphological analysis of neurons required for normal circadian behavior reveals subtle abnormalities suggesting that defects in axonal pathfinding or synapse formation may cause the observed behavioral defects.

Original language	English (US)
Pages (from-to)	973-984
Number of pages	12
Journal	Neuron
Volume	34
Issue number	6
DOIs	https://doi.org/10.1016/S0896-6273(02)00724-9
State	Published - Jun 13 2002
Externally published	Yes

All Science Journal Classification (ASJC) codes

Neuroscience(all)

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10.1016/S0896-6273(02)00724-9

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title = "Drosophila lacking dfmr1 activity show defects in circadian output and fail to maintain courtship interest",

abstract = "Fragile X mental retardation is a prominent genetic disorder caused by the lack of the FMR1 gene product a known RNA binding protein. Specific physiologic pathways regulated by FMR1 function have yet to be identified. Adult dfmr1 (also called dfxr) mutant flies display arrhythmic circadian activity and have erratic patterns of locomotor activity whereas overexpression of dFMR1 leads to a lengthened period. dfmr1 mutant males also display reduced courtship activity which appears to result from their inability to maintain courtship interest. Molecular analysis fails to reveal any defects in the expression of clock components; however the CREB output is affected. Morphological analysis of neurons required for normal circadian behavior reveals subtle abnormalities suggesting that defects in axonal pathfinding or synapse formation may cause the observed behavioral defects.",

author = "Dockendorff, {Thomas C.} and Su, {Henry S.} and McBride, {Sean M.J.} and Zhaohai Yang and Choi, {Catherine H.} and Siwicki, {Kathleen K.} and Amita Sehgal and Jongens, {Thomas A.}",

note = "Funding Information: We thank M. Fortini and H. Kazazian for critical comments on the manuscript. Special thanks go to the Sehgal laboratory for their discussions, interest, and help with experiments, especially Julie Williams, Edith Myers, and Sriram Sathyanarayanan. We would also like to thank Christian Klambt for futsch alleles. T.C.D. was supported in part by a postdoctoral fellowship from the FRAXA Foundation. This work was supported by NIH grants to T.A.J. and A.S. A.S. is an Associate Investigator of the Howard Hughes Medical Institute.",

year = "2002",

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doi = "10.1016/S0896-6273(02)00724-9",

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T1 - Drosophila lacking dfmr1 activity show defects in circadian output and fail to maintain courtship interest

AU - Dockendorff, Thomas C.

AU - Su, Henry S.

AU - McBride, Sean M.J.

AU - Yang, Zhaohai

AU - Choi, Catherine H.

AU - Siwicki, Kathleen K.

AU - Sehgal, Amita

AU - Jongens, Thomas A.

N1 - Funding Information: We thank M. Fortini and H. Kazazian for critical comments on the manuscript. Special thanks go to the Sehgal laboratory for their discussions, interest, and help with experiments, especially Julie Williams, Edith Myers, and Sriram Sathyanarayanan. We would also like to thank Christian Klambt for futsch alleles. T.C.D. was supported in part by a postdoctoral fellowship from the FRAXA Foundation. This work was supported by NIH grants to T.A.J. and A.S. A.S. is an Associate Investigator of the Howard Hughes Medical Institute.

PY - 2002/6/13

Y1 - 2002/6/13

N2 - Fragile X mental retardation is a prominent genetic disorder caused by the lack of the FMR1 gene product a known RNA binding protein. Specific physiologic pathways regulated by FMR1 function have yet to be identified. Adult dfmr1 (also called dfxr) mutant flies display arrhythmic circadian activity and have erratic patterns of locomotor activity whereas overexpression of dFMR1 leads to a lengthened period. dfmr1 mutant males also display reduced courtship activity which appears to result from their inability to maintain courtship interest. Molecular analysis fails to reveal any defects in the expression of clock components; however the CREB output is affected. Morphological analysis of neurons required for normal circadian behavior reveals subtle abnormalities suggesting that defects in axonal pathfinding or synapse formation may cause the observed behavioral defects.

AB - Fragile X mental retardation is a prominent genetic disorder caused by the lack of the FMR1 gene product a known RNA binding protein. Specific physiologic pathways regulated by FMR1 function have yet to be identified. Adult dfmr1 (also called dfxr) mutant flies display arrhythmic circadian activity and have erratic patterns of locomotor activity whereas overexpression of dFMR1 leads to a lengthened period. dfmr1 mutant males also display reduced courtship activity which appears to result from their inability to maintain courtship interest. Molecular analysis fails to reveal any defects in the expression of clock components; however the CREB output is affected. Morphological analysis of neurons required for normal circadian behavior reveals subtle abnormalities suggesting that defects in axonal pathfinding or synapse formation may cause the observed behavioral defects.

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Drosophila lacking dfmr1 activity show defects in circadian output and fail to maintain courtship interest

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