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Reversal of Disease-Related Pathologies in the Fragile X Mouse Model by Selective Activation of GABA B Receptors with Arbaclofen
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Pharmacological activation of the GABA B receptor with arbaclofen in a mouse model of fragile X syndrome corrects neuronal defects associated with the disease.
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In the citing paper
“Activation of the GABA B receptor by arbaclofen decreased mRNA translation in the cortex of fragile X mice and corrected the increased spine density 78 .”
From Progress toward treatments for synaptic defects in autism (2013) · cites this paper as [78]
In the citing paper
“Administration of GABAB agonists, such as baclofen or arbaclofen, corrects exacerbated protein synthesis and multiple phenotypes in Fmr1-KO mice37–39.”
From Drug development for neurodevelopmental disorders: lessons learned from fragile X syndrome (2018) · cites this paper as [37]
In the citing paper
“treatment with mGluR antagonists (mavoglurant) and GABAB agonists (arbaclofen), as well as gene reinstatement in Fmr1-KO mice starting in young adult or adult mice, can fully correct most behavioural, physiological, biochemical and neuroanatomical alterations30,32,37.”
From Drug development for neurodevelopmental disorders: lessons learned from fragile X syndrome (2018) · cites this paper as [37]
In the citing paper
“No human data are available for SCA1 in the absence of a drug suitable for clinical trials30,32,37.”
From Drug development for neurodevelopmental disorders: lessons learned from fragile X syndrome (2018) · cites this paper as [37]
In the citing paper
“For many subtypes of ASD, there is a GABAB deficit and arbaclofen has rescued the behavioral deficits including social deficits in the mouse models of idiopathic ASD [127], deletion of 16p11.2 [128], and FXS [129].”
From An Update on Psychopharmacological Treatment of Autism Spectrum Disorder (2022) · cites this paper as [129]
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Hagerman, R. J., Berry-Kravis, E., Hazlett, H. C., Bailey, D. B., Moine, H., Kooy, R. F., Tassone, F., Gantois, I., Sonenberg, N., Mandel, J. L., et al. (2017) · Nature Reviews Disease Primers · doi:10.1038/nrdp.2017.65 · Cited by 838
Jiang, Y.-H., Yuen, R. K. C., Jin, X., Wang, M., Chen, N., Wu, X., Ju, J., Mei, J., Shi, Y., He, M., et al. (2013) · The American Journal of Human Genetics · doi:10.1016/j.ajhg.2013.06.012 · Cited by 474
The translation of translational control by FMRP: therapeutic targets for FXS
Darnell, J. C., & Klann, E. (2013) · Nature Neuroscience · doi:10.1038/nn.3379 · Cited by 491
mTOR complexes in neurodevelopmental and neuropsychiatric disorders
Costa-Mattioli, M., & Monteggia, L. M. (2013) · Nature Neuroscience · doi:10.1038/nn.3546 · Cited by 367
Drug development for neurodevelopmental disorders: lessons learned from fragile X syndrome
Berry-Kravis, E. M., Lindemann, L., Jønch, A. E., Apostol, G., Bear, M. F., Carpenter, R. L., Crawley, J. N., Curie, A., Des Portes, V., Hossain, F., et al. (2018) · Nature Reviews Drug Discovery · doi:10.1038/nrd.2017.221 · Cited by 336
Self-citation
Metadata
- Authors
- Henderson, C., Wijetunge, L., Kinoshita, M. N., Shumway, M., Hammond, R. S., Postma, F. R., Brynczka, C., Rush, R., Thomas, A., Paylor, R., Warren, S. T., Vanderklish, P. W., Kind, P. C., Carpenter, R. L., Bear, M. F., & Healy, A. M.
- Affiliations
- Seaside Therapeutics (United States) (US)
- University of Edinburgh (GB)
- Emory University (US)
- Baylor College of Medicine (US)
- Scripps Research Institute (US)
- Massachusetts Institute of Technology (US)
- Corresponding author
- Healy, Aileen M.
- Venue
- Science Translational Medicine
- Published
- 2012-09-19
- Volume
- 4
- Issue
- 152
- Pages
- 152ra128-152ra128
- ISSN
- 1946-6234 (ISSN-L)
- 1946-6242
- PMC
- PMC8826584
- OpenAlex
- W2096270532
- Cited by
- 249
- References
- 102
- Citations by year
- 2026: 7 · 2025: 9 · 2024: 9 · 2023: 8 · 2022: 20 · 2021: 21 · 2020: 10 · 2019: 20 · 2018: 8 · 2017: 32 · 2016: 14 · 2015: 25 · 2014: 24 · 2013: 31 · 2012: 11
- FWCI
- 17.48
- Type
- journal article
- Language
- en
- Open access
- green
- www.ncbi.nlm.nih.gov
- Locations
- Science Translational Medicine (publishedVersion)
- PubMed (publishedVersion)
- PubMed Central (acceptedVersion)
- Funders
- Eunice Kennedy Shriver National Institute of Child Health and Human Development (HD 24064, HD020521, R01HD020521, P30HD024064, R37HD020521)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
- Life Sciences › Neuroscience › Cellular and Molecular Neuroscience › Neuroscience and Neuropharmacology Research
- Health Sciences › Medicine › Public Health, Environmental and Occupational Health › Down syndrome and intellectual disability research
- Keywords
- fragile X syndrome, GABAB receptors, baclofen, Fmr1 knockout mice, FMRP, synaptic protein synthesis, dendritic spine morphology, AMPA receptor internalization, mRNA translation, synaptic dysfunction, intellectual disability, autism spectrum disorder, hippocampus
- MeSH terms
- Animals, Baclofen, Behavior, Animal, Disease Models, Animal, Fragile X Syndrome, Hippocampus, Male, Phenotype, Polyribosomes, Seizures, Synapses, Protein Biosynthesis, Receptors, GABA-B, Receptors, AMPA, Mice, Knockout, Protein Transport, Dendritic Spines, Mice, Fragile X Messenger Ribonucleoprotein 1, GABA-B Receptor Agonists, Drinking Water
About these checks
Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.
Statistics and arithmetic are recomputed, not judged.
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