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.
authors.affiliation: Department of Genetics, University of Pennsylvania School of Medicine, Philadelphia, PA 19104, USA.
authors.forename: Thomas C
authors.initials: TC
authors.lastname: Dockendorff
authors.forename: Henry S
authors.initials: HS
authors.lastname: Su
authors.forename: Sean M J
authors.initials: SM
authors.lastname: McBride
authors.forename: Zhaohai
authors.initials: Z
authors.lastname: Yang
authors.forename: Catherine H
authors.initials: CH
authors.lastname: Choi
authors.forename: Kathleen K
authors.initials: KK
authors.lastname: Siwicki
authors.forename: Amita
authors.initials: A
authors.lastname: Sehgal
authors.forename: Thomas A
authors.initials: TA
authors.lastname: Jongens
mesh_terms: D000483:Alleles; D000818:Animals; D002940:Circadian Rhythm / Q000235:genetics* / Q000502:physiology; D003380:Courtship; D004330:Drosophila; D029721:Drosophila Proteins / Q000172:deficiency* / Q000235:genetics*; D005260:Female; D051860:Fragile X Messenger Ribonucleoprotein 1; D019476:Insect Proteins / Q000235:genetics / Q000502:physiology; D008297:Male; D008869:Microtubule-Associated Proteins / Q000235:genetics / Q000502:physiology; D009154:Mutation / Q000235:genetics*; D009414:Nerve Growth Factors / Q000235:genetics / Q000502:physiology; D009419:Nerve Tissue Proteins / Q000096:biosynthesis / Q000172:deficiency* / Q000235:genetics* / Q000502:physiology; D009687:Nuclear Proteins / Q000235:genetics / Q000502:physiology; D056950:Period Circadian Proteins; D016601:RNA-Binding Proteins
chemical_list: D029721:Drosophila Proteins; D051860:Fragile X Messenger Ribonucleoprotein 1; D019476:Insect Proteins; D008869:Microtubule-Associated Proteins; D009414:Nerve Growth Factors; D009419:Nerve Tissue Proteins; D009687:Nuclear Proteins; D056950:Period Circadian Proteins; D016601:RNA-Binding Proteins; C539040:PER protein, Drosophila; C409635:futsch protein, Drosophila; C096445:tim protein, Drosophila