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Modulation of dADAR-dependent RNA editing by the Drosophila fragile X mental retardation protein
by
Pepper, Anita S-R
, Jepson, James E
, Jongens, Thomas A
, Savva, Yiannis A
, Reenan, Robert A
, Bhogal, Balpreet
in
631/337/1645/1944
/ 631/378/1689
/ 631/378/2571/2577
/ Adenosine
/ Adenosine Deaminase - genetics
/ Adenosine Deaminase - metabolism
/ Analysis of Variance
/ Animal Genetics and Genomics
/ Animals
/ Animals, Genetically Modified
/ Antibodies
/ Behavioral Sciences
/ Biological Techniques
/ Biomedical and Life Sciences
/ Biomedicine
/ Cell Line, Transformed
/ Drosophila
/ Drosophila Proteins - genetics
/ Drosophila Proteins - metabolism
/ Editing
/ Fragile X Mental Retardation Protein - genetics
/ Fragile X Mental Retardation Protein - metabolism
/ Fragile X syndrome
/ Gene Expression Regulation, Developmental
/ Genetic aspects
/ Immunoprecipitation
/ Insects
/ Intellectual disabilities
/ Larva
/ Mass Spectrometry
/ Microscopy, Confocal
/ Mutation
/ Neurobiology
/ Neuromuscular Junction - cytology
/ Neuromuscular Junction - genetics
/ Neuromuscular Junction - metabolism
/ Neurosciences
/ Physiological aspects
/ Proteins
/ Risk factors
/ RNA
/ RNA Editing - genetics
/ RNA-Binding Proteins
/ Synaptic Transmission
/ Transfection
2011
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Modulation of dADAR-dependent RNA editing by the Drosophila fragile X mental retardation protein
by
Pepper, Anita S-R
, Jepson, James E
, Jongens, Thomas A
, Savva, Yiannis A
, Reenan, Robert A
, Bhogal, Balpreet
in
631/337/1645/1944
/ 631/378/1689
/ 631/378/2571/2577
/ Adenosine
/ Adenosine Deaminase - genetics
/ Adenosine Deaminase - metabolism
/ Analysis of Variance
/ Animal Genetics and Genomics
/ Animals
/ Animals, Genetically Modified
/ Antibodies
/ Behavioral Sciences
/ Biological Techniques
/ Biomedical and Life Sciences
/ Biomedicine
/ Cell Line, Transformed
/ Drosophila
/ Drosophila Proteins - genetics
/ Drosophila Proteins - metabolism
/ Editing
/ Fragile X Mental Retardation Protein - genetics
/ Fragile X Mental Retardation Protein - metabolism
/ Fragile X syndrome
/ Gene Expression Regulation, Developmental
/ Genetic aspects
/ Immunoprecipitation
/ Insects
/ Intellectual disabilities
/ Larva
/ Mass Spectrometry
/ Microscopy, Confocal
/ Mutation
/ Neurobiology
/ Neuromuscular Junction - cytology
/ Neuromuscular Junction - genetics
/ Neuromuscular Junction - metabolism
/ Neurosciences
/ Physiological aspects
/ Proteins
/ Risk factors
/ RNA
/ RNA Editing - genetics
/ RNA-Binding Proteins
/ Synaptic Transmission
/ Transfection
2011
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Modulation of dADAR-dependent RNA editing by the Drosophila fragile X mental retardation protein
by
Pepper, Anita S-R
, Jepson, James E
, Jongens, Thomas A
, Savva, Yiannis A
, Reenan, Robert A
, Bhogal, Balpreet
in
631/337/1645/1944
/ 631/378/1689
/ 631/378/2571/2577
/ Adenosine
/ Adenosine Deaminase - genetics
/ Adenosine Deaminase - metabolism
/ Analysis of Variance
/ Animal Genetics and Genomics
/ Animals
/ Animals, Genetically Modified
/ Antibodies
/ Behavioral Sciences
/ Biological Techniques
/ Biomedical and Life Sciences
/ Biomedicine
/ Cell Line, Transformed
/ Drosophila
/ Drosophila Proteins - genetics
/ Drosophila Proteins - metabolism
/ Editing
/ Fragile X Mental Retardation Protein - genetics
/ Fragile X Mental Retardation Protein - metabolism
/ Fragile X syndrome
/ Gene Expression Regulation, Developmental
/ Genetic aspects
/ Immunoprecipitation
/ Insects
/ Intellectual disabilities
/ Larva
/ Mass Spectrometry
/ Microscopy, Confocal
/ Mutation
/ Neurobiology
/ Neuromuscular Junction - cytology
/ Neuromuscular Junction - genetics
/ Neuromuscular Junction - metabolism
/ Neurosciences
/ Physiological aspects
/ Proteins
/ Risk factors
/ RNA
/ RNA Editing - genetics
/ RNA-Binding Proteins
/ Synaptic Transmission
/ Transfection
2011
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Modulation of dADAR-dependent RNA editing by the Drosophila fragile X mental retardation protein
Journal Article
Modulation of dADAR-dependent RNA editing by the Drosophila fragile X mental retardation protein
2011
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Overview
Loss of
FMR1
gene function results in fragile X syndrome. Here, the authors demonstrate that the
Drosophila
fragile X homolog dFMR1 is involved in the RNA editing pathway via interaction with the enzyme dADAR and suggest that proper NMJ synaptic architecture requires modulation of dADAR activity by dFMR1.
Loss of
FMR1
gene function results in fragile X syndrome, the most common heritable form of intellectual disability. The protein encoded by this locus (FMRP) is an RNA-binding protein that is thought to primarily act as a translational regulator; however, recent studies have implicated FMRP in other mechanisms of gene regulation. We found that the
Drosophila
fragile X homolog (dFMR1) biochemically interacted with the adenosine-to-inosine RNA-editing enzyme dADAR.
Adar
and
Fmr1
mutant larvae exhibited distinct morphological neuromuscular junction (NMJ) defects. Epistasis experiments based on these phenotypic differences revealed that
Adar
acts downstream of
Fmr1
and that dFMR1 modulates dADAR activity. Furthermore, sequence analyses revealed that a loss or overexpression of dFMR1 affects editing efficiency on certain dADAR targets with defined roles in synaptic transmission. These results link dFMR1 with the RNA-editing pathway and suggest that proper NMJ synaptic architecture requires modulation of dADAR activity by dFMR1.
Publisher
Nature Publishing Group US,Nature Publishing Group
Subject
/ Adenosine Deaminase - genetics
/ Adenosine Deaminase - metabolism
/ Animal Genetics and Genomics
/ Animals
/ Animals, Genetically Modified
/ Biomedical and Life Sciences
/ Drosophila Proteins - genetics
/ Drosophila Proteins - metabolism
/ Editing
/ Fragile X Mental Retardation Protein - genetics
/ Fragile X Mental Retardation Protein - metabolism
/ Gene Expression Regulation, Developmental
/ Insects
/ Larva
/ Mutation
/ Neuromuscular Junction - cytology
/ Neuromuscular Junction - genetics
/ Neuromuscular Junction - metabolism
/ Proteins
/ RNA
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