Fmr1

Summary

Gene Symbol: Fmr1
Description: Fmr1
Alias: AT24755, BcDNA:GM08679, CG6203, DFMRP, Dmel\CG6203, EP(3)3517, FMR, FMR1, FMRP, FMRp, FXR, Fmrp, cg6203, dFMR, dFMR1, dFMRP, dFXR, dFXR1, dFXRP, dFmr1, dFmrp, dfmr, dfmr1, dfxr, dfxr1, dmfr1, fmr, fmr1, Fmr1, CG6203-PA, CG6203-PB, CG6203-PC, CG6203-PD, CG6203-PE, CG6203-PF, CG6203-PG, CG6203-PH, CG6203-PI, CG6203-PJ, CG6203-PK, Fmr1-PA, Fmr1-PB, Fmr1-PC, Fmr1-PD, Fmr1-PE, Fmr1-PF, Fmr1-PG, Fmr1-PH, Fmr1-PI, Fmr1-PJ, Fmr1-PK, Fragile-X mental retardation 1, Fragile-X mental retardation protein, Fragile-X-related, dFMR, dFmr1, drosophila fragile X mental retardation protein, fragile X, fragile X mental retardation, fragile X mental retardation 1, fragile X mental retardation gene, fragile X mental retardation protein, fragile X mental retardation protein 1, fragile X protein, fragile X related protein, fragile X-related, fragile x related, fragile-X
Species: fruit fly
Products:     Fmr1

Top Publications

  1. Sofola O, Sundram V, Ng F, Kleyner Y, Morales J, Botas J, et al. The Drosophila FMRP and LARK RNA-binding proteins function together to regulate eye development and circadian behavior. J Neurosci. 2008;28:10200-5 pubmed publisher
    ..FXS patients have a deficit for the fragile X mental retardation protein (FMRP) that results in abnormal neuronal dendritic spine morphology and behavioral phenotypes, ..
  2. Gatto C, Broadie K. Fragile X mental retardation protein is required for programmed cell death and clearance of developmentally-transient peptidergic neurons. Dev Biol. 2011;356:291-307 pubmed publisher
    Fragile X syndrome (FXS), caused by loss of fragile X mental retardation 1 (FMR1) gene function, is the most common heritable cause of intellectual disability and autism spectrum disorders...
  3. D Hulst C, De Geest N, Reeve S, Van Dam D, De Deyn P, Hassan B, et al. Decreased expression of the GABAA receptor in fragile X syndrome. Brain Res. 2006;1121:238-45 pubmed
    ..wide screening for differentially expressed mRNAs in brain of fragile X mice, a validated model for fragile X mental retardation syndrome, we analyzed expression of the 17 remaining subunits of the GABA(A) receptor using real-time ..
  4. Okamura K, Ishizuka A, Siomi H, Siomi M. Distinct roles for Argonaute proteins in small RNA-directed RNA cleavage pathways. Genes Dev. 2004;18:1655-66 pubmed
    ..Our findings show that distinct Argonaute proteins act at different steps of the small RNA silencing mechanism and suggest that there are inherent differences between siRNA-initiated RISCs and miRNA-initiated RISCs in Drosophila. ..
  5. Bhogal B, Jepson J, Savva Y, Pepper A, Reenan R, Jongens T. Modulation of dADAR-dependent RNA editing by the Drosophila fragile X mental retardation protein. Nat Neurosci. 2011;14:1517-24 pubmed publisher
    Loss of FMR1 gene function results in fragile X syndrome, the most common heritable form of intellectual disability...
  6. Schenck A, Van de Bor V, Bardoni B, Giangrande A. Novel features of dFMR1, the Drosophila orthologue of the fragile X mental retardation protein. Neurobiol Dis. 2002;11:53-63 pubmed
    ..The Drosophila genome contains a single gene homologous to the FXR family. We show that dFMR1 is subjected to transcriptional and posttranscriptional regulation during development and that it homomerizes, ..
  7. Schenck A, Bardoni B, Langmann C, Harden N, Mandel J, Giangrande A. CYFIP/Sra-1 controls neuronal connectivity in Drosophila and links the Rac1 GTPase pathway to the fragile X protein. Neuron. 2003;38:887-98 pubmed
    ..Rho GTPase pathways control actin reorganization, while the fragile X mental retardation protein (FMRP) regulates the synthesis of specific proteins...
  8. Cziko A, McCann C, Howlett I, Barbee S, Duncan R, Luedemann R, et al. Genetic modifiers of dFMR1 encode RNA granule components in Drosophila. Genetics. 2009;182:1051-60 pubmed publisher
    ..The Fragile-X Mental Retardation protein (FMRP/dFMR1) is one of the best-studied neuronal translational control molecules and here we describe the identification and ..
  9. Pan L, Broadie K. Drosophila fragile X mental retardation protein and metabotropic glutamate receptor A convergently regulate the synaptic ratio of ionotropic glutamate receptor subclasses. J Neurosci. 2007;27:12378-89 pubmed
    A current hypothesis proposes that fragile X mental retardation protein (FMRP), an RNA-binding translational regulator, acts downstream of glutamatergic transmission, via metabotropic glutamate receptor (mGluR) G(q)-dependent signaling, ..

More Information

Publications76

  1. Siomi M, Higashijima K, Ishizuka A, Siomi H. Casein kinase II phosphorylates the fragile X mental retardation protein and modulates its biological properties. Mol Cell Biol. 2002;22:8438-47 pubmed
    b>Fragile X syndrome is caused by loss of FMR1 protein expression. FMR1 binds RNA and associates with polysomes in the cytoplasm; thus, it has been proposed to function as a regulator of gene expression at the posttranscriptional level...
  2. Bettencourt Da Cruz A, Schwarzel M, Schulze S, Niyyati M, Heisenberg M, Kretzschmar D. Disruption of the MAP1B-related protein FUTSCH leads to changes in the neuronal cytoskeleton, axonal transport defects, and progressive neurodegeneration in Drosophila. Mol Biol Cell. 2005;16:2433-42 pubmed
    ..Futsch is negatively regulated by the Drosophila Fragile X mental retardation gene, and a mutation in this gene delays the onset of neurodegeneration in futsch(olk)...
  3. Bolduc F, Bell K, Cox H, Broadie K, Tully T. Excess protein synthesis in Drosophila fragile X mutants impairs long-term memory. Nat Neurosci. 2008;11:1143-5 pubmed publisher
    ..We observed that fragile X protein was acutely required and interacted with argonaute1 and staufen in the formation of long-term memory...
  4. Darnell J, Fraser C, Mostovetsky O, Darnell R. Discrimination of common and unique RNA-binding activities among Fragile X mental retardation protein paralogs. Hum Mol Genet. 2009;18:3164-77 pubmed publisher
    b>Fragile X mental retardation is caused by loss-of-function of a single gene encoding FMRP, an RNA-binding protein that harbors three canonical RNA-binding domains, two KH-type and one RGG box...
  5. Kaytor M, Orr H. RNA targets of the fragile X protein. Cell. 2001;107:555-7 pubmed
    ..in Cell bring the power of human genetics, Drosophila genetics, and genomics to bear on the understanding of fragile X syndrome...
  6. Barbee S, Estes P, Cziko A, Hillebrand J, Luedeman R, Coller J, et al. Staufen- and FMRP-containing neuronal RNPs are structurally and functionally related to somatic P bodies. Neuron. 2006;52:997-1009 pubmed
    ..Here, we show that staufen- and FMRP-containing RNPs in Drosophila neurons contain proteins also present in somatic "P bodies," including the ..
  7. Pan L, Zhang Y, Woodruff E, Broadie K. The Drosophila fragile X gene negatively regulates neuronal elaboration and synaptic differentiation. Curr Biol. 2004;14:1863-70 pubmed
    ..The disease is caused by the silencing of the fragile X mental retardation 1 (fmr1) gene, which encodes the RNA binding translational regulator FMRP ...
  8. Estes P, O Shea M, Clasen S, Zarnescu D. Fragile X protein controls the efficacy of mRNA transport in Drosophila neurons. Mol Cell Neurosci. 2008;39:170-9 pubmed publisher
    b>Fragile X syndrome, the most common form of inherited mental retardation is caused by mutations in the FMR1 gene. FMR1 encodes an RNA-binding protein thought to control the transport and translation of target mRNAs...
  9. Epstein A, Bauer C, Ho A, Bosco G, Zarnescu D. Drosophila Fragile X protein controls cellular proliferation by regulating cbl levels in the ovary. Dev Biol. 2009;330:83-92 pubmed publisher
    FMRP is an RNA binding protein linked to the most common form of inherited mental retardation, Fragile X syndrome (FraX)...
  10. Bushey D, Tononi G, Cirelli C. The Drosophila fragile X mental retardation gene regulates sleep need. J Neurosci. 2009;29:1948-61 pubmed publisher
    ..The fragile X mental retardation gene Fmr1, whose loss-of-function mutation causes the most common form of inherited mental retardation in ..
  11. Chang S, Bray S, Li Z, Zarnescu D, He C, Jin P, et al. Identification of small molecules rescuing fragile X syndrome phenotypes in Drosophila. Nat Chem Biol. 2008;4:256-63 pubmed publisher
    Fragile X syndrome is caused by the functional loss of the fragile X mental retardation 1 (FMR1) gene. Deletion of the FMR1 ortholog in Drosophila melanogaster (Fmr1) recapitulates many phenotypes associated with fragile X syndrome...
  12. Costa A, Wang Y, Dockendorff T, Erdjument Bromage H, Tempst P, Schedl P, et al. The Drosophila fragile X protein functions as a negative regulator in the orb autoregulatory pathway. Dev Cell. 2005;8:331-42 pubmed
    ..Here we show that Orb associates with the Drosophila Fragile X Mental Retardation (dFMR1) protein as part of a ribonucleoprotein complex that controls the localized translation of mRNAs ..
  13. Michel C, Kraft R, Restifo L. Defective neuronal development in the mushroom bodies of Drosophila fragile X mental retardation 1 mutants. J Neurosci. 2004;24:5798-809 pubmed
    b>Fragile X mental retardation 1 (Fmr1) is a highly conserved gene with major roles in CNS structure and function...
  14. Zarnescu D, Jin P, Betschinger J, Nakamoto M, Wang Y, Dockendorff T, et al. Fragile X protein functions with lgl and the par complex in flies and mice. Dev Cell. 2005;8:43-52 pubmed
    ..syndrome, the most common form of inherited mental retardation, is caused by loss of function for the Fragile X Mental Retardation 1 gene (FMR1)...
  15. Caudy A, Myers M, Hannon G, Hammond S. Fragile X-related protein and VIG associate with the RNA interference machinery. Genes Dev. 2002;16:2491-6 pubmed
    ..We identify two putative RNA-binding proteins, the Drosophila homolog of the fragile X mental retardation protein (FMRP), dFXR, and VIG (Vasa intronic gene), through their association with RISC...
  16. Tessier C, Broadie K. Drosophila fragile X mental retardation protein developmentally regulates activity-dependent axon pruning. Development. 2008;135:1547-57 pubmed publisher
    ..Loss of the fragile X mental retardation 1 (fmr1) gene product, the mRNA-binding translational regulator FMRP, causes structural over-elaboration ..
  17. Du T, Zamore P. microPrimer: the biogenesis and function of microRNA. Development. 2005;132:4645-52 pubmed
    ..miRNAs function very much like siRNAs, but these two types of small RNAs can be distinguished by their distinct pathways for maturation and by the logic by which they regulate gene expression. ..
  18. Kavi H, Fernandez H, Xie W, Birchler J. RNA silencing in Drosophila. FEBS Lett. 2005;579:5940-9 pubmed
    ..RNA silencing processes in Drosophila are described. ..
  19. Ishizuka A, Siomi M, Siomi H. A Drosophila fragile X protein interacts with components of RNAi and ribosomal proteins. Genes Dev. 2002;16:2497-508 pubmed
    b>Fragile X syndrome is a common form of inherited mental retardation caused by the loss of FMR1 expression...
  20. Reeve S, Lin X, Sahin B, Jiang F, Yao A, Liu Z, et al. Mutational analysis establishes a critical role for the N terminus of fragile X mental retardation protein FMRP. J Neurosci. 2008;28:3221-6 pubmed publisher
    ..is the most common form of heritable mental retardation caused by the loss of function of the fragile X mental retardation protein FMRP...
  21. Callan M, Clements N, Ahrendt N, Zarnescu D. Fragile X Protein is required for inhibition of insulin signaling and regulates glial-dependent neuroblast reactivation in the developing brain. Brain Res. 2012;1462:151-61 pubmed publisher
    b>Fragile X syndrome (FXS) is the most common form of inherited mental disability and known cause of autism...
  22. Zhang Y, Friedman D, Wang Z, Woodruff E, Pan L, O DONNELL J, et al. Protein expression profiling of the drosophila fragile X mutant brain reveals up-regulation of monoamine synthesis. Mol Cell Proteomics. 2005;4:278-90 pubmed
    ..The disease is caused by silencing of the fragile X mental retardation 1 (fmr1) gene, which encodes the mRNA-binding, translational regulator FMRP...
  23. Beerman R, Jongens T. A non-canonical start codon in the Drosophila fragile X gene yields two functional isoforms. Neuroscience. 2011;181:48-66 pubmed publisher
    Fragile X syndrome is caused by the loss of expression of the fragile X mental retardation protein (FMRP)...
  24. Monzo K, Papoulas O, Cantin G, Wang Y, Yates J, Sisson J. Fragile X mental retardation protein controls trailer hitch expression and cleavage furrow formation in Drosophila embryos. Proc Natl Acad Sci U S A. 2006;103:18160-5 pubmed
    ..Here, we demonstrate that Drosophila fragile X mental retardation protein (dFMRP) is required for cleavage furrow formation and functions within dynamic cytoplasmic ..
  25. Callan M, Cabernard C, Heck J, Luois S, Doe C, Zarnescu D. Fragile X protein controls neural stem cell proliferation in the Drosophila brain. Hum Mol Genet. 2010;19:3068-79 pubmed publisher
    ..FXS) is the most common form of inherited mental retardation and is caused by the loss of function for Fragile X protein (FMRP), an RNA-binding protein thought to regulate synaptic plasticity by controlling the localization and ..
  26. Choi C, McBride S, Schoenfeld B, Liebelt D, Ferreiro D, Ferrick N, et al. Age-dependent cognitive impairment in a Drosophila fragile X model and its pharmacological rescue. Biogerontology. 2010;11:347-62 pubmed publisher
    ..This demonstrates a role for the Drosophila Fragile X Mental Retardation Protein (dFMR1) in neuronal physiology with regard to cognition during the aging process...
  27. Martinez V, Javadi C, Ngo E, Ngo L, Lagow R, Zhang B. Age-related changes in climbing behavior and neural circuit physiology in Drosophila. Dev Neurobiol. 2007;67:778-91 pubmed
  28. Repicky S, Broadie K. Metabotropic glutamate receptor-mediated use-dependent down-regulation of synaptic excitability involves the fragile X mental retardation protein. J Neurophysiol. 2009;101:672-87 pubmed publisher
    ..protein FMRP results in the most common inherited form of both mental retardation and autism spectrum disorders: fragile X syndrome (FXS)...
  29. Pan L, Woodruff E, Liang P, Broadie K. Mechanistic relationships between Drosophila fragile X mental retardation protein and metabotropic glutamate receptor A signaling. Mol Cell Neurosci. 2008;37:747-60 pubmed publisher
    b>Fragile X syndrome is caused by loss of the FMRP translational regulator. A current hypothesis proposes that FMRP functions downstream of mGluR signaling to regulate synaptic connections...
  30. Tessier C, Broadie K. The fragile X mental retardation protein developmentally regulates the strength and fidelity of calcium signaling in Drosophila mushroom body neurons. Neurobiol Dis. 2011;41:147-59 pubmed publisher
    ..FXS is caused by loss of the fragile X mental retardation 1 (FMR1) gene, whose FMRP product regulates mRNA translation downstream of synaptic activity to modulate ..
  31. Zhang Y, Bailey A, Matthies H, Renden R, Smith M, Speese S, et al. Drosophila fragile X-related gene regulates the MAP1B homolog Futsch to control synaptic structure and function. Cell. 2001;107:591-603 pubmed
    b>Fragile X mental retardation gene (FMR1) encodes an RNA binding protein that acts as a negative translational regulator...
  32. Pepper A, Beerman R, Bhogal B, Jongens T. Argonaute2 suppresses Drosophila fragile X expression preventing neurogenesis and oogenesis defects. PLoS ONE. 2009;4:e7618 pubmed publisher
    Fragile X Syndrome is caused by the silencing of the Fragile X Mental Retardation gene (FMR1)...
  33. McBride S, Choi C, Wang Y, Liebelt D, Braunstein E, Ferreiro D, et al. Pharmacological rescue of synaptic plasticity, courtship behavior, and mushroom body defects in a Drosophila model of fragile X syndrome. Neuron. 2005;45:753-64 pubmed
    b>Fragile X syndrome is a leading heritable cause of mental retardation that results from the loss of FMR1 gene function...
  34. Siller S, Broadie K. Matrix metalloproteinases and minocycline: therapeutic avenues for fragile X syndrome. Neural Plast. 2012;2012:124548 pubmed publisher
    b>Fragile X syndrome (FXS) is the most common known genetic form of intellectual disability and autism spectrum disorders...
  35. Lee A, Li W, Xu K, Bogert B, Su K, Gao F. Control of dendritic development by the Drosophila fragile X-related gene involves the small GTPase Rac1. Development. 2003;130:5543-52 pubmed
    Fragile X syndrome is caused by loss-of-function mutations in the fragile X mental retardation 1 gene. How these mutations affect neuronal development and function remains largely elusive...
  36. Friedman S, Dani N, Rushton E, Broadie K. Fragile X mental retardation protein regulates trans-synaptic signaling in Drosophila. Dis Model Mech. 2013;6:1400-13 pubmed publisher
    ..determinant of intellectual disability and autism spectrum disorders, is caused by loss of the fragile X mental retardation 1 (FMR1) gene product (FMRP), an mRNA-binding translational repressor...
  37. Papoulas O, Monzo K, Cantin G, Ruse C, Yates J, Ryu Y, et al. dFMRP and Caprin, translational regulators of synaptic plasticity, control the cell cycle at the Drosophila mid-blastula transition. Development. 2010;137:4201-9 pubmed publisher
    ..b>Drosophila Fragile X mental retardation protein (dFMRP; FMR1), a transcript-specific translational regulator, is required for cellularization...
  38. Billuart P, Chelly J. From fragile X mental retardation protein to Rac1 GTPase: new insights from Fly CYFIP. Neuron. 2003;38:843-5 pubmed
    Mutations in either the Rho GTPase pathway or in the fragile X mental retardation (FMR1) gene produce neuronal connectivity defects. In this issue of Neuron, Schenck et al...
  39. Sekine T, Yamaguchi T, Hamano K, Siomi H, Saez L, Ishida N, et al. Circadian phenotypes of Drosophila fragile x mutants in alternative genetic backgrounds. Zoolog Sci. 2008;25:561-71 pubmed publisher
    Drosophila FMR1 mutants are models of human fragile X syndrome. They show a loss of locomotor activity rhythm and severe degradation of eclosion timing...
  40. Banerjee P, Nayar S, Hebbar S, Fox C, Jacobs M, Park J, et al. Substitution of critical isoleucines in the KH domains of Drosophila fragile X protein results in partial loss-of-function phenotypes. Genetics. 2007;175:1241-50 pubmed
    b>Fragile X mental retardation proteins (FMRP) are RNA-binding proteins that interact with a subset of cellular RNAs...
  41. Yang L, Duan R, Chen D, Wang J, Chen D, Jin P. Fragile X mental retardation protein modulates the fate of germline stem cells in Drosophila. Hum Mol Genet. 2007;16:1814-20 pubmed
    Fragile X syndrome, a common form of inherited mental retardation, is caused by the loss of fragile X mental retardation protein (FMRP)...
  42. Jin P, Zarnescu D, Ceman S, Nakamoto M, Mowrey J, Jongens T, et al. Biochemical and genetic interaction between the fragile X mental retardation protein and the microRNA pathway. Nat Neurosci. 2004;7:113-7 pubmed
    Fragile X syndrome is caused by a loss of expression of the fragile X mental retardation protein (FMRP). FMRP is a selective RNA-binding protein which forms a messenger ribonucleoprotein (mRNP) complex that associates with polyribosomes...
  43. Gatto C, Broadie K. Temporal requirements of the fragile x mental retardation protein in modulating circadian clock circuit synaptic architecture. Front Neural Circuits. 2009;3:8 pubmed publisher
    Loss of fragile X mental retardation 1 (FMR1) gene function is the most common cause of inherited mental retardation and autism spectrum disorders, characterized by attention disorder, hyperactivity and disruption of circadian activity ..
  44. Kirkpatrick L, McIlwain K, Nelson D. Comparative genomic sequence analysis of the FXR gene family: FMR1, FXR1, and FXR2. Genomics. 2001;78:169-77 pubmed
    Mutations in the X-linked gene FMR1 cause fragile X syndrome, the leading cause of inherited mental retardation. Two autosomal paralogs of FMR1 have been identified, and are known as FXR1 and FXR2...
  45. van Alphen B, Yap M, Kirszenblat L, Kottler B, van Swinderen B. A dynamic deep sleep stage in Drosophila. J Neurosci. 2013;33:6917-27 pubmed publisher
    ..Our results show that sleep is not homogenous in insects, and suggest that waking behavior and the associated synaptic plasticity mechanisms determine the timing and intensity of deep sleep stages in Drosophila...
  46. Xu X, Li Y, Wang F, Gao F. The steady-state level of the nervous-system-specific microRNA-124a is regulated by dFMR1 in Drosophila. J Neurosci. 2008;28:11883-9 pubmed publisher
    Fragile X syndrome is the most common form of inherited mental retardation caused by loss of the fragile X mental retardation protein 1 (FMRP)...
  47. Wan L, Dockendorff T, Jongens T, Dreyfuss G. Characterization of dFMR1, a Drosophila melanogaster homolog of the fragile X mental retardation protein. Mol Cell Biol. 2000;20:8536-47 pubmed
    b>Fragile X syndrome is the most common inherited form of mental retardation. It is caused by loss of FMR1 gene activity due to either lack of expression or expression of a mutant form of the protein...
  48. Farny N, Kedersha N, Silver P. Metazoan stress granule assembly is mediated by P-eIF2alpha-dependent and -independent mechanisms. RNA. 2009;15:1814-21 pubmed publisher
    ..These results suggest that mammals evolved alternative mechanisms for dealing with thermal stress. ..
  49. Mines M, Jope R. Glycogen synthase kinase-3: a promising therapeutic target for fragile x syndrome. Front Mol Neurosci. 2011;4:35 pubmed publisher
    ..FXS results from inadequate expression of functional fragile X mental retardation protein (FMRP)...
  50. Ling S, Fahrner P, Greenough W, Gelfand V. Transport of Drosophila fragile X mental retardation protein-containing ribonucleoprotein granules by kinesin-1 and cytoplasmic dynein. Proc Natl Acad Sci U S A. 2004;101:17428-33 pubmed
    ..b>Fragile X mental retardation protein (FMRP) has been shown to be involved in translational regulation and is found in ribonucleoprotein (..
  51. Stetler A, Winograd C, Sayegh J, Cheever A, Patton E, Zhang X, et al. Identification and characterization of the methyl arginines in the fragile X mental retardation protein Fmrp. Hum Mol Genet. 2006;15:87-96 pubmed
    b>Fragile X syndrome is the most common form of inherited mental retardation and is caused by the absence of expression of the FMR1 gene...
  52. Gao F. Understanding fragile X syndrome: insights from retarded flies. Neuron. 2002;34:859-62 pubmed
    ..the most common form of inherited mental retardation, is caused by loss-of-function mutations in the fragile X mental retardation 1 (fmr1) gene...
  53. Kelley D, Davidson R, Elliott J, Lahvis G, Yin J, Bhattacharyya A. The cyclic AMP cascade is altered in the fragile X nervous system. PLoS ONE. 2007;2:e931 pubmed
    ..FX results from a trinucleotide expansion mutation in the fmr1 gene that reduces levels of fragile X mental retardation protein (FMRP)...
  54. Coffee R, Tessier C, Woodruff E, Broadie K. Fragile X mental retardation protein has a unique, evolutionarily conserved neuronal function not shared with FXR1P or FXR2P. Dis Model Mech. 2010;3:471-85 pubmed publisher
    Fragile X syndrome (FXS), resulting solely from the loss of function of the human fragile X mental retardation 1 (hFMR1) gene, is the most common heritable cause of mental retardation and autism disorders, with syndromic defects also in ..
  55. Dockendorff T, Su H, McBride S, Yang Z, Choi C, Siwicki K, et al. Drosophila lacking dfmr1 activity show defects in circadian output and fail to maintain courtship interest. Neuron. 2002;34:973-84 pubmed
    b>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...
  56. Gatto C, Broadie K. Temporal requirements of the fragile X mental retardation protein in the regulation of synaptic structure. Development. 2008;135:2637-48 pubmed publisher
    b>Fragile X syndrome (FraX), caused by the loss-of-function of one gene (FMR1), is the most common inherited form of both mental retardation and autism spectrum disorders...
  57. Kim K, Lee Y, Carthew R. Conversion of pre-RISC to holo-RISC by Ago2 during assembly of RNAi complexes. RNA. 2007;13:22-9 pubmed
    ..We have also identified a new intermediate complex in the RISC assembly pathway, pre-RISC, in which Ago2 is stably bound to double-stranded siRNA. ..
  58. Yao A, Jin S, Li X, Liu Z, Ma X, Tang J, et al. Drosophila FMRP regulates microtubule network formation and axonal transport of mitochondria. Hum Mol Genet. 2011;20:51-63 pubmed publisher
    ..X syndrome, the most common form of inherited mental retardation, is caused by the absence of the fragile X mental retardation protein FMRP...
  59. Siller S, Broadie K. Neural circuit architecture defects in a Drosophila model of Fragile X syndrome are alleviated by minocycline treatment and genetic removal of matrix metalloproteinase. Dis Model Mech. 2011;4:673-85 pubmed publisher
    Fragile X syndrome (FXS), caused by loss of the fragile X mental retardation 1 (FMR1) product (FMRP), is the most common cause of inherited intellectual disability and autism spectrum disorders...
  60. Bushey D, Tononi G, Cirelli C. Sleep and synaptic homeostasis: structural evidence in Drosophila. Science. 2011;332:1576-81 pubmed publisher
    ..A richer wake experience resulted in both larger synaptic growth and greater sleep need. Finally, we demonstrate that the gene Fmr1 (fragile X mental retardation 1) plays an important role in sleep-dependent synaptic renormalization.
  61. Banerjee P, Schoenfeld B, Bell A, Choi C, Bradley M, Hinchey P, et al. Short- and long-term memory are modulated by multiple isoforms of the fragile X mental retardation protein. J Neurosci. 2010;30:6782-92 pubmed publisher
    ..b>Fragile X mental retardation protein (FMRP), a neuronal RNA binding protein, exists in isoforms as a result of alternative splicing, but ..
  62. Xu K, Bogert B, Li W, Su K, Lee A, Gao F. The fragile X-related gene affects the crawling behavior of Drosophila larvae by regulating the mRNA level of the DEG/ENaC protein pickpocket1. Curr Biol. 2004;14:1025-34 pubmed
    Fragile X syndrome is caused by loss-of-function mutations in the fragile X mental retardation 1 (FMR1) gene. How FMR1 affects the function of the central and peripheral nervous systems is still unclear...
  63. Deshpande G, Calhoun G, Schedl P. The drosophila fragile X protein dFMR1 is required during early embryogenesis for pole cell formation and rapid nuclear division cycles. Genetics. 2006;174:1287-98 pubmed
    ..In humans, inactivation of the X-linked FMR gene fragile X is the most common cause of mental retardation and leads to defects in neuronal architecture...
  64. Inoue S, Shimoda M, Nishinokubi I, Siomi M, Okamura M, Nakamura A, et al. A role for the Drosophila fragile X-related gene in circadian output. Curr Biol. 2002;12:1331-5 pubmed
    Mutations that abolish expression of an X-linked gene, FMR1, result in the pathogenesis of fragile X syndrome, the most common form of inherited mental retardation...
  65. Kanellopoulos A, Semelidou O, Kotini A, Anezaki M, Skoulakis E. Learning and memory deficits consequent to reduction of the fragile X mental retardation protein result from metabotropic glutamate receptor-mediated inhibition of cAMP signaling in Drosophila. J Neurosci. 2012;32:13111-24 pubmed
    Loss of the RNA-binding fragile X protein [fragile X mental retardation protein (FMRP)] results in a spectrum of cognitive deficits, the fragile X syndrome (FXS), while aging individuals with decreased protein levels present with a subset ..
  66. Yang Y, Xu S, Xia L, Wang J, Wen S, Jin P, et al. The bantam microRNA is associated with drosophila fragile X mental retardation protein and regulates the fate of germline stem cells. PLoS Genet. 2009;5:e1000444 pubmed publisher
    Fragile X syndrome, a common form of inherited mental retardation, is caused by the loss of fragile X mental retardation protein (FMRP)...
  67. Morales J, Hiesinger P, Schroeder A, Kume K, Verstreken P, Jackson F, et al. Drosophila fragile X protein, DFXR, regulates neuronal morphology and function in the brain. Neuron. 2002;34:961-72 pubmed
    Mental retardation is a pervasive societal problem, 25 times more common than blindness for example. Fragile X syndrome, the most common form of inherited mental retardation, is caused by mutations in the FMR1 gene...