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1 FMRP did not appear to regulate synapses individually, b
2 FMRP has been confirmed to bind voltage-gated potassium
3 FMRP is a master regulator of local translation but its
4 FMRP is a RNA-binding protein predominantly resident in
5 FMRP is a selective RNA binding protein owing to two cen
6 FMRP is an mRNA-binding protein regulating neuronal tran
7 FMRP is an RNA-binding protein involved in the control o
8 FMRP is an RNA-binding protein that is involved in the t
9 FMRP is both an RNA- and channel-binding regulator, with
10 FMRP is present predominantly in the cytoplasm, where it
11 FMRP uses an arginine-glycine-rich (RGG) motif for speci
12 FMRP was also shown to interact with the auxiliary beta4
13 FMRP was shown to directly interact with, and modulate,
14 FMRP-null PNs lose activity-dependent synaptic modulatio
15 FMRP-null PNs reduce synaptic branching and enlarge bout
16 e of fragile X mental retardation protein 1 (FMRP) leads to both pre- and postsynaptic defects, yet w
17 nucleotide repeat expansions in FMR1 abolish FMRP expression, leading to hyperactivation of ERK and m
19 behavioral plasticity occurs normally after FMRP knockdown, but performance rapidly deteriorated in
22 out of FMRP suppresses, and expression of an FMRP mutant protein that fails to interact with Cdh1 phe
23 tural modifications and learning triggers an FMRP-dependent increase of alphaCaMKII dendritic transla
28 led a novel mechanism by which Gp1 mGluR and FMRP mediate protein translation and neural network acti
29 this issue, we measured brain Fmr1 mRNA and FMRP levels as a function of CGG-repeat length in a cong
31 t Mdm2 acts as a translation suppressor, and FMRP is required for its ubiquitination and down-regulat
33 that PFC dysfunction may persist as long as FMRP is absent and therefore can be rescued after develo
35 idence that the selective loss of astroglial FMRP contributes to cortical synaptic deficits in FXS, l
36 s show that the selective loss of astroglial FMRP contributes to cortical synaptic deficits in FXS, p
37 ns; whether the selective loss of astroglial FMRP in vivo alters astrocyte functions and contributes
38 Although the selective loss of astroglial FMRP only modestly increases spine density and length in
42 advances focus on discovering links between FMRP roles to determine whether FMRP has a multitude of
46 es have attempted to identify mRNAs bound by FMRP through several methods, each generating a list of
52 coordinated regulation of PSD95/Dlg4 mRNA by FMRP and FXR2P that ultimately affects its fine-tuning d
53 st that specific binding of cellular RNAs by FMRP may involve hydrogen bonding with RNA duplexes and
56 tal retardation 1 protein (Fmr1, also called FMRP) acts independently of futsch/MAP-1B to abolish act
62 microarray analyses of endfeet, we discover FMRP-bound transcripts, which encode signaling and cytos
67 ous Cdh1-APC forms a complex with endogenous FMRP, and knockout of Cdh1 impairs mGluR-induced ubiquit
68 it remains unclear, however, to what extent FMRP-BK channel interactions contribute to synaptic and
71 oportionately longer lengths, enrichment for FMRP binding and G-quartets, and their genes are under g
72 rate an independent presynaptic function for FMRP through the study of an ID patient with an FMR1 mis
73 ut direct demonstration of a requirement for FMRP control of local protein synthesis during behaviora
74 ing synaptoneurosomes demonstrate a role for FMRP and miR-125a in regulating the translation of PSD-9
77 n the Drosophila FXS disease model, we found FMRP binds shrub mRNA (human Chmp4) to repress Shrub exp
79 ntral brain learning/memory center, we found FMRP loss and Shrub overexpression similarly increase co
84 d from birth through adulthood, with greater FMRP reductions in the soma than in the neurite, despite
85 expansion (preCGG), cortical and hippocampal FMRP expression is moderately reduced from birth through
90 l structure of the complex between the human FMRP RGG peptide bound to the in vitro selected G-rich R
91 ator and chicken are highly similar to human FMRP with identical mRNA-binding and phosphorylation sit
92 the FCBS dataset of reproducibly identified FMRP binding sites is a valuable tool for investigating
94 atment with the tPA-neutralizing antibody in FMRP-deficient cells during early neural progenitor diff
97 we observed highly clustered WGGA motifs in FMRP targets compared with other genes, implicating both
98 ent behavioral plasticity occurs normally in FMRP knockdown animals, but plasticity degrades over 24
100 This study may provide a novel insight into FMRP involvement in the intracellular localization of FU
104 circuit, we discovered that neurons lacking FMRP take up dramatically more current-injected small dy
105 physical property of central neurons lacking FMRP that could underlie aspects of FXS disruption of ne
109 mediated transcription regulation and mGluR5/FMRP-mediated protein translation regulation through cor
110 17 FXTAS individuals revealed that the mild FMRP decrease in mice mirrored the more limited data for
111 a, and are enriched for chromatin modifiers, FMRP-associated genes and embryonically expressed genes.
113 , neuronally driven expression of the mutant FMRP is unable to rescue structural defects at the neuro
114 osophila FXS model is key to discovering new FMRP roles, because of its genetic malleability and indi
116 rn neurons that are prevented by ablation of FMRP in adult-born neurons and rescued by an metabotropi
117 y bulb neurons and cell-specific ablation of FMRP, we investigated whether learning shapes adult-born
118 gulate BK channel activity in the absence of FMRP and determine its ability to normalize multilevel d
119 an be attributed to the continued absence of FMRP from the PFC, independent of FMRP status during dev
121 these deficits (1) are due to the absence of FMRP in the PFC alone and (2) are not the result of deve
122 dysfunction due to the continued absence of FMRP is necessary to understand the different roles of F
131 arget mRNAs, we produced a shared dataset of FMRP consensus binding sequences (FCBS), which were repr
134 ccurs between the carboxy-terminal domain of FMRP and domains of Ca(V)2.2 known to interact with the
137 yramidal neurons, selective re-expression of FMRP in astrocytes significantly attenuates abnormal spi
139 tic- and translation-independent function of FMRP that is linked to a specific subset of FXS phenotyp
140 ether the pre- and postsynaptic functions of FMRP are independent and have distinct roles in FXS neur
141 tudies identified important new functions of FMRP in regulating neural excitability and synaptic tran
143 but the molecular mechanism and identity of FMRP targets mediating this phenotype remain largely unk
146 more, we confirmed the direct interaction of FMRP with one candidate, RhoC, by in vitro RNA binding a
147 endent mechanisms and direct interactions of FMRP with a number of ion channels in the axons and pres
148 (Cyfip2) was identified as an interactor of FMRP, and its mRNA is a highly ranked FMRP target in mou
151 minent cell groups expressing high levels of FMRP at the subcortical levels, in particular sensory an
152 r brain areas contain high and low levels of FMRP cell groups adjacent to each other or between layer
157 stinct from the current RNA-binding model of FMRP, we show that FMRP occupies the GAR domain of TRF2-
160 gether, these findings support the notion of FMRP differential neuronal regulation and strongly impli
161 ortant, we describe differential patterns of FMRP distribution in both cortical and subcortical brain
162 sive experience decreases phosphorylation of FMRP in the NAc, which is coupled to a long-term increas
164 cits are rescued by initiating production of FMRP in adult conditional restoration mice, suggesting t
167 ogether reveal an unexpected nuclear role of FMRP in DDR and uncover a feed-forward mechanism by whic
170 cessary to understand the different roles of FMRP and to treat patients effectively throughout life.
171 at it is possible to dissociate the roles of FMRP in neural function from developmental dysregulation
174 the well-characterized target transcripts of FMRP are synaptic proteins, yet targeting these proteins
175 uR impairs activity-dependent translation of FMRP, which may hinder synaptic plasticity in a clinical
179 k show that elevated PN activity phenocopies FMRP-null defects, whereas PN silencing causes opposing
182 in NL and in particular a list of potential FMRP targets, with the goal of facilitating molecular ch
183 This mutation, c.413G > A (R138Q), preserves FMRP's canonical functions in RNA binding and translatio
184 nels and mechanisms that mediate presynaptic FMRP actions, it remains unclear, however, to what exten
187 eins, including the syndromic autism protein FMRP, move in basal processes at velocities consistent w
190 ene silencing and insufficient FMR1 protein (FMRP), FXTAS is thought to be caused by 'toxicity' of ex
194 oss of fragile X mental retardation protein (FMRP) and haploinsufficiency of synaptic GTPase-activati
195 oss of fragile X mental retardation protein (FMRP) and that FMRP acts on BK channels by modulating th
197 oss of fragile X mental retardation protein (FMRP) cause fragile X syndrome (FXS), a genetic disorder
199 ion of fragile X mental retardation protein (FMRP) causes fragile X syndrome (FXS) have largely focus
200 oss of fragile X mental retardation protein (FMRP) causes fragile X syndrome (FXS), yet the mechanism
201 nce of Fragile X Mental Retardation Protein (FMRP) from birth results in developmental disabilities a
202 oss of fragile X mental retardation protein (FMRP) in different brain cell types, especially in non-n
206 The fragile X mental retardation protein (FMRP) is an mRNA binding protein that regulates activity
208 oss of fragile X mental retardation protein (FMRP) is thought to underlie cognitive deficits in FXS,
209 nce of fragile X mental retardation protein (FMRP) leads to defects in plasticity and learning defici
212 of the fragile X mental retardation protein (FMRP) pathway (10 observed vs. 4.4 expected, P = .0076).
213 gh the fragile X mental retardation protein (FMRP) pathway may underlie synaptic plasticity associate
214 The fragile X mental retardation protein (FMRP) plays an important role in normal brain developmen
215 AD) of fragile X mental retardation protein (FMRP) protein is considered to be a member of the methyl
216 s, the Fragile X Mental Retardation protein (FMRP) regulates expression of the scaffolding postsynapt
217 The Fragile X mental retardation protein (FMRP) regulates neuronal RNA metabolism, and its absence
219 ion of fragile X mental retardation protein (FMRP), a highly selective RNA-binding protein and transl
221 of the fragile X mental retardation protein (FMRP), an RNA binding protein that regulates translation
222 le for fragile X mental retardation protein (FMRP), an RNA-binding protein and regulator of dendritic
223 ently, fragile X mental retardation protein (FMRP), an RNA-binding protein that regulates local prote
224 genous fragile X mental retardation protein (FMRP), and a reporter containing a patient 3'UTR caused
225 n with Fragile X mental retardation protein (FMRP), its upregulation in transformed lymphoblastoid ce
226 ent on fragile X mental retardation protein (FMRP), the protein that is deficient in the most common
228 ion of fragile X mental retardation protein (FMRP), which represses translation of target transcripts
238 of the fragile X mental retardation protein (FMRP, product of FMR1) are enriched for case mutations.
240 y and interacts with an RNA-binding protein, FMRP, to promote synapse formation; and Top3beta gene de
241 ore of the Fragile X related (FXR) proteins (FMRP, FXR2P, and FXR1P) along with mRNA and ribosomes.
243 d divergent sets of target mRNA and putative FMRP-bound motifs, and a clear understanding of FMRP's b
249 the loss of the mRNA translational repressor FMRP leads to exaggerated protein synthesis downstream o
255 al motifs that have been proposed to specify FMRP binding, the short sequence motifs TGGA and GAC wer
256 X mental retardation protein (FMRP) and that FMRP acts on BK channels by modulating the channel's gat
258 but no direct evidence has demonstrated that FMRP-regulated dendritic protein synthesis affects behav
259 stribution of the FCBS set demonstrates that FMRP preferentially binds to the coding region of its ta
260 ll, Chen et al. (2014) provide evidence that FMRP represses translation by binding the ribosome, sugg
263 These differential patterns indicate that FMRP expression appears to be specific to individual neu
266 immunoprecipitation in cortical neurons that FMRP is mostly associated with one unique mRNA: diacylgl
271 rent RNA-binding model of FMRP, we show that FMRP occupies the GAR domain of TRF2-S protein to block
272 Experiments in Fmr1 null mice show that FMRP regulates axonal protein expression but is not requ
280 in BLAST analyses first demonstrate that the FMRP amino acid sequences in the alligator and chicken a
282 translation to investigate the role of this FMRP messenger RNA target in learning-dependent structur
285 ino terminal crystal structures of wild-type FMRP, and a mutant (R138Q) that disrupts the amino termi
287 s to FXS pathology and support a model where FMRP, by controlling the translation of Dgkkappa, indire
288 local protein synthesis in vivo and whether FMRP knockdown affects protein synthesis-dependent visua
289 inks between FMRP roles to determine whether FMRP has a multitude of unrelated functions or whether c
291 n effort to determine the mechanism by which FMRP mediates protein translation and neural network act
292 nes, leading to distinct hypotheses by which FMRP recognizes its targets; namely, by RNA structure or
293 ndividuals are defective in association with FMRP; whereas one of the mutants is also deficient in bi
294 on-related protein 2 (FXR2P) cooperates with FMRP in binding to the 3'-UTR of mouse PSD95/Dlg4 mRNA.
295 sensor mechanism based on sensory cues, with FMRP loss causing the most common heritable autism spect
297 ion channel shown to directly interact with FMRP; this interaction alters the single-channel propert
298 researchers established the causal link with FMRP loss >;25 years ago, studies continue to reveal div
299 teins might form collaborative networks with FMRP and possibly other post-transcriptional regulators
300 ce, that these significantly overlapped with FMRP direct targets and/or SFARI human autism genes, and
301 redominantly express a single FXG type, with FMRP-containing FXGs the most prevalent in forebrain neu
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