Drosophila lacking dfmr1 activity show defects in circadian output and fail to maintain courtship interest - PubMed (original) (raw)
Drosophila lacking dfmr1 activity show defects in circadian output and fail to maintain courtship interest
Thomas C Dockendorff et al. Neuron. 2002.
Free article
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.
Similar articles
- Drosophila fragile X protein, DFXR, regulates neuronal morphology and function in the brain.
Morales J, Hiesinger PR, Schroeder AJ, Kume K, Verstreken P, Jackson FR, Nelson DL, Hassan BA. Morales J, et al. Neuron. 2002 Jun 13;34(6):961-72. doi: 10.1016/s0896-6273(02)00731-6. Neuron. 2002. PMID: 12086643 - A role for the Drosophila fragile X-related gene in circadian output.
Inoue S, Shimoda M, Nishinokubi I, Siomi MC, Okamura M, Nakamura A, Kobayashi S, Ishida N, Siomi H. Inoue S, et al. Curr Biol. 2002 Aug 6;12(15):1331-5. doi: 10.1016/s0960-9822(02)01036-9. Curr Biol. 2002. PMID: 12176363 - A circadian output in Drosophila mediated by neurofibromatosis-1 and Ras/MAPK.
Williams JA, Su HS, Bernards A, Field J, Sehgal A. Williams JA, et al. Science. 2001 Sep 21;293(5538):2251-6. doi: 10.1126/science.1063097. Science. 2001. PMID: 11567138 - Understanding fragile X syndrome: insights from retarded flies.
Gao FB. Gao FB. Neuron. 2002 Jun 13;34(6):859-62. doi: 10.1016/s0896-6273(02)00740-7. Neuron. 2002. PMID: 12086633 Review. - RNA interference: a new mechanism by which FMRP acts in the normal brain? What can Drosophila teach us?
Siomi H, Ishizuka A, Siomi MC. Siomi H, et al. Ment Retard Dev Disabil Res Rev. 2004;10(1):68-74. doi: 10.1002/mrdd.20011. Ment Retard Dev Disabil Res Rev. 2004. PMID: 14994291 Review.
Cited by
- Activation of the 5-HT1A Receptor by Eltoprazine Restores Mitochondrial and Motor Deficits in a Drosophila Model of Fragile X Syndrome.
Vannelli A, Mariano V, Bagni C, Kanellopoulos AK. Vannelli A, et al. Int J Mol Sci. 2024 Aug 13;25(16):8787. doi: 10.3390/ijms25168787. Int J Mol Sci. 2024. PMID: 39201473 Free PMC article. - Circadian Rhythm and Sleep Analyses in a Fruit Fly Model of Fragile X Syndrome Using a Video-Based Automated Behavioral Research System.
Milojevic S, Ghosh A, Makevic V, Stojkovic M, Capovilla M, Tosti T, Budimirovic D, Protic D. Milojevic S, et al. Int J Mol Sci. 2024 Jul 20;25(14):7949. doi: 10.3390/ijms25147949. Int J Mol Sci. 2024. PMID: 39063191 Free PMC article. - From wings to whiskers to stem cells: why every model matters in fragile X syndrome research.
Sandoval SO, Méndez-Albelo NM, Xu Z, Zhao X. Sandoval SO, et al. J Neurodev Disord. 2024 Jun 13;16(1):30. doi: 10.1186/s11689-024-09545-w. J Neurodev Disord. 2024. PMID: 38872088 Free PMC article. Review. - PGC-1α integrates insulin signaling with mitochondrial physiology and behavior in a Drosophila model of Fragile X Syndrome.
Weisz ED, Fenton AR, Jongens TA. Weisz ED, et al. NPJ Metab Health Dis. 2024;2:2. doi: 10.1038/s44324-024-00004-7. Epub 2024 Feb 21. NPJ Metab Health Dis. 2024. PMID: 38741938 Free PMC article. - Tet controls axon guidance in early brain development through glutamatergic signaling.
Tran H, Le L, Singh BN, Kramer J, Steward R. Tran H, et al. iScience. 2024 Apr 1;27(5):109634. doi: 10.1016/j.isci.2024.109634. eCollection 2024 May 17. iScience. 2024. PMID: 38655199 Free PMC article.
Publication types
MeSH terms
Substances
LinkOut - more resources
Full Text Sources
Other Literature Sources
Molecular Biology Databases