Using Drosophila as a tool to identify pharmacological therapies for fragile X syndrome

Drug Discovery Today: Technologies

Volumn 10, Issue 1, 2013, Pages

  McBride, Sean M J ^a   Holloway, Sandra L ^a   Jongens, Thomas A ^a  

^a UNIVERSITY OF PENNSYLVANIA   (United States)

Author keywords

[No Author keywords available]

Indexed keywords

2 AMINO 2 (2 CARBOXYCYCLOPROPYL) 3 (XANTHEN 9 YL)PROPIONIC ACID; 2 METHYL 6 (PHENYLETHYNYL)PYRIDINE; BENZATROPINE; FRAGILE X MENTAL RETARDATION PROTEIN; METABOTROPIC RECEPTOR; MINOCYCLINE; NIPECOTIC ACID; PILOCARPINE; RESERPINE;

BEHAVIOR; CELL DIVISION; CELL GROWTH; CELL METABOLISM; COGNITION; DROSOPHILA MELANOGASTER; FRAGILE X SYNDROME; GENE MUTATION; LEARNING; MEMORY; NEUROANATOMY; NONHUMAN; PHENOTYPE; REVIEW; SEQUENCE HOMOLOGY;

ANIMALS; DISEASE MODELS, ANIMAL; DROSOPHILA; DRUG EVALUATION, PRECLINICAL; FRAGILE X SYNDROME; HUMANS;

EID: 84875757666     PISSN: None     EISSN: 17406749     Source Type: Journal    
DOI: 10.1016/j.ddtec.2012.09.005     Document Type: Review

References (83)

1. S. Jacquemont Fragile-X syndrome and fragile X-associated tremor/ataxia syndrome: two faces of FMR1 Lancet Neurol. 6 2007 45 55
2. R. Hagerman Fragile X syndrome and targeted treatment trials Results Probl. Cell Differ. 54 2012 297 335
3. S.M. McBride Behavior in a Drosophila model of fragile X Results Probl. Cell Differ. 54 2012 83 117
4. C.R. Tessier, and K. Broadie Molecular and genetic analysis of the Drosophila model of fragile X syndrome Results Probl. Cell Differ. 54 2012 119 156
5. W.T. O'Donnell, and S.T. Warren A decade of molecular studies of fragile X syndrome Annu. Rev. Neurosci. 25 2002 315 338
6. D.D. Hatton Autistic behavior in children with fragile X syndrome: prevalence, stability, and the impact of FMRP Am. J. Med. Genet. A 140A 2006 1804 1813
7. D.I. Zafeiriou Childhood autism and associated comorbidities Brain Dev. 29 2007 257 272
8. J.S. Sutcliffe DNA methylation represses FMR-1 transcription in fragile X syndrome Hum. Mol. Genet. 1 1992 397 400
9. A.J. Verkerk Identification of a gene (FMR-1) containing a CGG repeat coincident with a breakpoint cluster region exhibiting length variation in fragile X syndrome Cell 65 1991 905 914
10. C.T. Ashley Jr. FMR1 protein: conserved RNP family domains and selective RNA binding Science 262 1993 563 566
11. L. Wan Characterization of dFMR1: a Drosophila melanogaster homolog of the fragile X mental retardation protein Mol. Cell. Biol. 20 2000 8536 8547
12. J.C. Darnell Discrimination of common and unique RNA-binding activities among fragile X mental retardation protein paralogs Hum. Mol. Genet. 18 2009 3164 3177
13. Y.Q. Zhang Drosophila fragile X-related gene regulates the MAP1B homolog Futsch to control synaptic structure and function Cell 107 2001 591 603
14. J. Morales Drosophila fragile X protein: DFXR, regulates neuronal morphology and function in the brain Neuron 34 2002 961 972
15. T.C. Dockendorff Drosophila lacking dfmr1 activity show defects in circadian output and fail to maintain courtship interest Neuron 34 2002 973 984
16. S. Inoue A role for the Drosophila fragile X-related gene in circadian output Curr. Biol.: CB 12 2002 1331 1335
17. S.M. McBride Pharmacological rescue of synaptic plasticity: courtship behavior, and mushroom body defects in a Drosophila model of fragile X syndrome Neuron 45 2005 753 764
18. G. Dolen, and M.F. Bear Courting a cure for fragile X Neuron 45 2005 642 644
19. B. Bhogal, and T.A. Jongens Fragile X syndrome and model organisms: identifying potential routes of therapeutic intervention Dis. Model. Mech. 3 11-12 2010 693 700
20. E. Berry-Kravis Open-label treatment trial of lithium to target the underlying defect in fragile X syndrome J. Dev. Behav. Pediatr. 29 4 2008 293 302
21. S. Jacquemont Epigenetic modification of the FMR1 gene in fragile X syndrome is associated with differential response to the mGluR5 antagonist AFQ056 Sci. Transl. Med. 3 64 2011 64ra1
22. Q.J. Yan Suppression of two major fragile X syndrome mouse model phenotypes by the mGluR5 antagonist MPEP Neuropharmacology 49 7 2005 1053 1066
23. G. Dolen Correction of fragile X syndrome in mice Neuron 56 6 2007 955 962
24. S.S. Siller, and K. Broadie 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. 4 5 2011 673 685
25. T.V. Bilousova Minocycline promotes dendritic spine maturation and improves behavioural performance in the fragile X mouse model J. Med. Genet. 46 2 2009 94 102
26. C.J. Yuskaitis Lithium ameliorates altered glycogen synthase kinase-3 and behavior in a mouse model of fragile X syndrome Biochem. Pharmacol. 79 4 2010 632 646
27. W.W. Min Elevated glycogen synthase kinase-3 activity in fragile X mice: key metabolic regulator with evidence for treatment potential Neuropharmacology 56 2 2009 463 472
28. B.J. Dickson Wired for sex: the neurobiology of Drosophila mating decisions Science 322 5903 2008 904 909
29. K.K. Siwicki, and L. Ladewski Associative learning and memory in Drosophila: beyond olfactory conditioning Behav. Process. 64 2 2003 225 238
30. L.C. Griffith, and A. Ejima Courtship learning in Drosophila melanogaster: diverse plasticity of a reproductive behavior Learn. Mem. 16 12 2009 743 750
31. J.M. Tauber Elevated levels of the vesicular monoamine transporter and a novel repetitive behavior in the Drosophila model of fragile X syndrome PLoS One 6 11 2011 e27100
32. W.G. Quinn Conditioned behavior in Drosophila melanogaster Proc. Natl. Acad. Sci. U. S. A. 71 3 1974 708 712
33. Y. Dudai dunce: a mutant of Drosophila deficient in learning Proc. Natl. Acad. Sci. U. S. A. 73 5 1976 1684 1688
34. J.A. Jellies Associative Olfactory Conditioning in Drosophila melanogaster and Memory Retention Through Metamorphosis 1981 Illinois State University p. 83
35. T. Tully, and W.G. Quinn Classical conditioning and retention in normal and mutant Drosophila melanogaster J. Comp. Physiol. A 157 2 1985 263 277
36. R.L. Davis Mushroom bodies and Drosophila learning Neuron 11 1 1993 1 14
37. T. Tully Genetic dissection of consolidated memory in Drosophila Cell 79 1 1994 35 47
38. E.M. Skoulakis, and S. Grammenoudi Dunces and da Vincis: the genetics of learning and memory in Drosophila Cell. Mol. Life Sci. 63 9 2006 975 988
39. F.V. Bolduc Excess protein synthesis in Drosophila fragile X mutants impairs long-term memory Nat. Neurosci. 11 10 2008 1143 1145
40. R.W. Siegel, and J.C. Hall Conditioned responses in courtship behavior of normal and mutant Drosophila Proc. Natl. Acad. Sci. U. S. A. 76 7 1979 3430 3434
41. J.C. Hall The mating of a fly Science 264 5166 1994 1702 1714
42. J.C. Hall Control of male reproductive behavior by the central nervous system of Drosophila: dissection of a courtship pathway by genetic mosaics Genetics 92 2 1979 437 457
43. L. Tompkins Conditioned courtship in Drosophila and its mediation by association of chemical cues Behav. Genet. 13 6 1983 565 578
44. L. Tompkins The role of female movement in the sexual behavior of Drosophila melanogaster Behav. Genet. 12 3 1982 295 307
45. L. Tompkins Genetic analysis of sex appeal in Drosophila Behav. Genet. 14 5 1984 411 440
46. S.L. Ackerman, and R.W. Siegel Chemically reinforced conditioned courtship in Drosophila: responses of wild-type and the dunce, amnesiac and don giovanni mutants J. Neurogenet. 3 2 1986 111 123
47. K.K. Siwicki The role of cuticular pheromones in courtship conditioning of Drosophila males Learn. Mem. 12 6 2005 636 645
48. A. Ejima Sequential learning of pheromonal cues modulates memory consolidation in trainer-specific associative courtship conditioning Curr. Biol. 15 3 2005 194 206
49. A. Ejima Generalization of courtship learning in Drosophila is mediated by cis-vaccenyl acetate Curr. Biol. 17 7 2007 599 605
50. S.M. McBride Mushroom body ablation impairs short-term memory and long-term memory of courtship conditioning in Drosophila melanogaster Neuron 24 4 1999 967 977
51. C.H. Choi Age-dependent cognitive impairment in a Drosophila fragile X model and its pharmacological rescue Biogerontology 11 3 2010 347 362
52. P. Banerjee Short- and long-term memory are modulated by multiple isoforms of the fragile X mental retardation protein J. Neurosci. 30 19 2010 6782 6792
53. C.X. He, and C. Portera-Cailliau The trouble with spines in fragile X syndrome: density, maturity and plasticity Neuroscience 2012
54. L.L. Restifo Mental retardation genes in Drosophila: new approaches to understanding and treating developmental brain disorders Ment. Retard. Dev. Disabil. Res. Rev. 11 4 2005 286 294
55. L. Pan The Drosophila fragile X gene negatively regulates neuronal elaboration and synaptic differentiation Curr. Biol. 14 20 2004 1863 1870
56. J.C. Yin Induction of a dominant negative CREB transgene specifically blocks long-term memory in Drosophila Cell 79 1 1994 49 58
57. E. Berry-Kravis, and P.R. Huttenlocher Cyclic AMP metabolism in fragile X syndrome Ann. Neurol. 31 1 1992 22 26
58. M.J. Berridge Inositol trisphosphate and calcium signalling Nature 361 6410 1993 315 325
59. P.L. Lakin-Thomas Effects of inositol starvation on the levels of inositol phosphates and inositol lipids in Neurospora crassa Biochem. J. 292 Pt 3 1993 805 811
60. E. Berry-Kravis Reduced cyclic AMP production in fragile X syndrome: cytogenetic and molecular correlations Pediatr. Res. 38 5 1995 638 643
61. E. Berry-Kravis, and R. Ciurlionis Overexpression of fragile X gene (FMR-1) transcripts increases cAMP production in neural cells J. Neurosci. Res. 51 1 1998 41 48
62. B.P. Bullock, and J.F. Habener Phosphorylation of the cAMP response element binding protein CREB by cAMP-dependent protein kinase A and glycogen synthase kinase-3 alters DNA-binding affinity: conformation, and increases net charge Biochemistry 37 11 1998 3795 3809
63. T. Inoue Type 1 inositol 1,4,5-trisphosphate receptor is required for induction of long-term depression in cerebellar Purkinje neurons J. Neurosci. 18 14 1998 5366 5373
64. K. Takei Regulation of nerve growth mediated by inositol 1,4,5-trisphosphate receptors in growth cones Science 282 5394 1998 1705 1708
65. M.F. Bear The mGluR theory of fragile X mental retardation Trends Neurosci. 27 7 2004 370 377
66. K.M. Huber Altered synaptic plasticity in a mouse model of fragile X mental retardation Proc. Natl. Acad. Sci. U. S. A. 99 11 2002 7746 7750
67. C.H. Choi Pharmacological reversal of synaptic plasticity deficits in the mouse model of fragile X syndrome by group II mGluR antagonist or lithium treatment Brain Res. 1380 2011 106 119
68. L. Pan, and K.S. Broadie Drosophila fragile X mental retardation protein and metabotropic glutamate receptor A convergently regulate the synaptic ratio of ionotropic glutamate receptor subclasses J. Neurosci. 27 45 2007 12378 12389
69. L. Pan Mechanistic relationships between Drosophila fragile X mental retardation protein and metabotropic glutamate receptor A signaling Mol. Cell. Neurosci. 37 4 2008 747 760
70. S.O. Moldin Understanding fragile X syndrome: molecular, cellular and genomic neuroscience at the crossroads Genes Brain Behav. 4 6 2005 337 340
71. S.O. Moldin Can autism speak to neuroscience? J. Neurosci.: Off. J. Soc. Neurosci. 26 26 2006 6893 6896
72. C.A. Walsh Autism and brain development Cell 135 3 2008 396 400
73. F.R. Volkmar Autism and autism spectrum disorders: diagnostic issues for the coming decade J. Child Psychol. Psychiatry Allied Discipl. 50 1-2 2009 108 115
74. F.L. Raymond, and P. Tarpey The genetics of mental retardation Hum. Mol. Genet. 15 2006 R110 R116
75. M.W. State The genetics of child psychiatric disorders: focus on autism and Tourette syndrome Neuron 68 2 2010 254 269
76. A. Michalon Chronic pharmacological mGlu5 inhibition corrects fragile X in adult mice Neuron 74 1 2012 49 56
77. S. Chang Identification of small molecules rescuing fragile X syndrome phenotypes in Drosophila Nat. Chem. Biol. 4 4 2008 256 263
78. I. Gantois Expression profiling suggests underexpression of the GABA(A) receptor subunit delta in the fragile X knockout mouse model Neurobiol. Dis. 21 2 2006 346 357
79. A.J. Sanchez Rolipram impairs NF-kappaB activity and MMP-9 expression in experimental autoimmune encephalomyelitis J. Neuroimmunol. 168 1-2 2005 13 20
80. C.A. Martin-Chouly Modulation of matrix metalloproteinase production from human lung fibroblasts by type 4 phosphodiesterase inhibitors Life Sci. 75 7 2004 823 840
81. S. Oger Evidence for a role of phosphodiesterase 4 in lipopolysaccharide-stimulated prostaglandin E2 production and matrix metalloproteinase-9 activity in human amniochorionic membranes J. Immunol. 174 12 2005 8082 8089
82. W. Liu Effect of glutamine on the expression of grp75 in PC12 cells Shi Yan Sheng Wu Xue Bao 38 5 2005 423 431
83. C. Paribello Open-label add-on treatment trial of minocycline in fragile X syndrome BMC Neurol. 10 2010 91