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

Neuron. 2002 Jun 13;34(6):973-84. doi: 10.1016/s0896-6273(02)00724-9.

Authors

Thomas C Dockendorff¹, Henry S Su, Sean M J McBride, Zhaohai Yang, Catherine H Choi, Kathleen K Siwicki, Amita Sehgal, Thomas A Jongens

Affiliation

¹ Department of Genetics, University of Pennsylvania School of Medicine, Philadelphia, PA 19104, USA.

PMID: 12086644
DOI: 10.1016/s0896-6273(02)00724-9

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.

Publication types

Research Support, Non-U.S. Gov't
Research Support, U.S. Gov't, P.H.S.

MeSH terms

Alleles
Animals
Circadian Rhythm / genetics*
Circadian Rhythm / physiology
Courtship*
Drosophila
Drosophila Proteins / deficiency*
Drosophila Proteins / genetics*
Female
Fragile X Mental Retardation Protein
Insect Proteins / genetics
Insect Proteins / physiology
Male
Microtubule-Associated Proteins / genetics
Microtubule-Associated Proteins / physiology
Mutation / genetics*
Nerve Growth Factors / genetics
Nerve Growth Factors / physiology
Nerve Tissue Proteins / biosynthesis
Nerve Tissue Proteins / deficiency*
Nerve Tissue Proteins / genetics*
Nerve Tissue Proteins / physiology
Nuclear Proteins / genetics
Nuclear Proteins / physiology
Period Circadian Proteins
RNA-Binding Proteins*

Substances

Drosophila Proteins
Insect Proteins
Microtubule-Associated Proteins
Nerve Growth Factors
Nerve Tissue Proteins
Nuclear Proteins
PER protein, Drosophila
Period Circadian Proteins
RNA-Binding Proteins
futsch protein, Drosophila
tim protein, Drosophila
Fragile X Mental Retardation Protein