ReviewDisease models & mechanisms
Fragile X syndrome and model organisms: identifying potential routes of therapeutic intervention.
Review in Disease models & mechanisms. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 20 papers.
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
20 citing papers in PubMed.
- From wings to whiskers to stem cells: why every model matters in fragile X syndrome research.Journal of neurodevelopmental disorders · 2024Review
- PGC-1α integrates insulin signaling with mitochondrial physiology and behavior in a Drosophila model of Fragile X Syndrome.npj metabolic health and disease · 2024Article
- Article
- Hyperexcitability and Homeostasis in Fragile X Syndrome.Frontiers in molecular neuroscience · 2021Article
- Wnd/DLK Is a Critical Target of FMRP Responsible for Neurodevelopmental and Behavior Defects in the Drosophila Model of Fragile X Syndrome.Cell reports · 2019Article
- Autism sensory dysfunction in an evolutionarily conserved system.Proceedings. Biological sciences · 2018Article
- Loss of Drosophila FMRP leads to alterations in energy metabolism and mitochondrial function.Human molecular genetics · 2018Article
- PDE-4 inhibition rescues aberrant synaptic plasticity in Drosophila and mouse models of fragile X syndrome.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2015Article
- RNA-binding protein misregulation in microsatellite expansion disorders.Advances in experimental medicine and biology · 2014Review
- Involvement of Rho-family GTPases in axon branching.Small GTPases · 2014Review
- Lithium treatment alleviates impaired cognition in a mouse model of fragile X syndrome.Genes, brain, and behavior · 2013Article
- A cell-based fascin bioassay identifies compounds with potential anti-metastasis or cognition-enhancing functions.Disease models & mechanisms · 2013Article
- UsingDrug discovery today. Technologies · 2012Article
- Synaptic dysfunction in neurodevelopmental disorders associated with autism and intellectual disabilities.Cold Spring Harbor perspectives in biology · 2012Review
- In vivo neuronal function of the fragile X mental retardation protein is regulated by phosphorylation.Human molecular genetics · 2012Article
- Networking in autism: leveraging genetic, biomarker and model system findings in the search for new treatments.Neuropsychopharmacology : official publication of the American College of Neuropsychopharmacology · 2012Review
- Therapeutic strategies in fragile X syndrome: dysregulated mGluR signaling and beyond.Neuropsychopharmacology : official publication of the American College of Neuropsychopharmacology · 2012Review
- Drosophila modeling of heritable neurodevelopmental disorders.Current opinion in neurobiology · 2011Review
- Fragile X mental retardation protein is required for programmed cell death and clearance of developmentally-transient peptidergic neurons.Developmental biology · 2011Article
- Article
Corrections and comments
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Authors and funding
2 authors.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Fragile X syndrome (FXS) is a cognitive disorder caused by silencing of the fragile X mental retardation 1 gene (FMR1). Since the discovery of the gene almost two decades ago, most scientific contributions have focused on identifying the molecular function of the fragile X mental retardation protein (FMRP) and understanding how absence of FMR1 gene expression gives rise to the disease phenotypes. The use of model organisms has allowed rapid progression in the FXS field and has given insight into the molecular basis of the disease. The mouse and fly FXS models have enabled studies to identify potential targets and pathways for pharmacological treatment. Here, we briefly review the two primary FXS model systems and describe how studies in these organisms have led us closer to therapeutic treatments for patients afflicted with FXS.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.