ArticleDevelopmental biology2011
Fragile X mental retardation protein is required for programmed cell death and clearance of developmentally-transient peptidergic neurons.
Article in Developmental biology, 2011. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 21 papers.
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Who cites it
21 citing papers in PubMed, 32 citations in OpenAlex.
- A rapid and dynamic role for FMRP in the plasticity of adult neurons.Nature communications · 2025Article
- Tango-seq: overlaying transcriptomics on connectomics to identify neurons downstream ofbioRxiv : the preprint server for biology · 2024Article
- Experience-dependent glial pruning of synaptic glomeruli during the critical period.Scientific reports · 2024Article
- Chemokine-like Orion is involved in the transformation of glial cells into phagocytes in different developmental neuronal remodeling paradigms.Development (Cambridge, England) · 2023Article
- Fragile X mental retardation protein coordinates neuron-to-glia communication for clearance of developmentally transient brain neurons.Proceedings of the National Academy of Sciences of the United States of America · 2023Article
- Dysregulation of BMP, Wnt, and Insulin Signaling in Fragile X Syndrome.Frontiers in cell and developmental biology · 2022Review
- Retinoic Acid Supplementation Rescues the Social Deficits inFrontiers in genetics · 2022Article
- Neuronal fragile X mental retardation protein activates glial insulin receptor mediated PDF-Tri neuron developmental clearance.Nature communications · 2021Article
- Genetic background mutations drive neural circuit hyperconnectivity in a fragile X syndrome model.BMC biology · 2020Article
- Disrupting flight increases sleep and identifies a novel sleep-promoting pathway inScience advances · 2020Article
- Unraveling the Pathways to Neuronal Homeostasis and Disease: Mechanistic Insights into the Role of RNA-Binding Proteins and Associated Factors.International journal of molecular sciences · 2018Review
- Modeling Congenital Disorders of N-Linked Glycoprotein Glycosylation inFrontiers in genetics · 2018Review
- Modeling Fragile X Syndrome inFrontiers in molecular neuroscience · 2018Review
- ESCRT-III Membrane Trafficking Misregulation Contributes To Fragile X Syndrome Synaptic Defects.Scientific reports · 2017Article
- miR-124 Regulates the Phase of Drosophila Circadian Locomotor Behavior.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2016Article
- Deciphering discord: How Drosophila research has enhanced our understanding of the importance of FMRP in different spatial and temporal contexts.Experimental neurology · 2015Review
- Activity-dependent FMRP requirements in development of the neural circuitry of learning and memory.Development (Cambridge, England) · 2015Article
- GABAergic circuit dysfunction in the Drosophila Fragile X syndrome model.Neurobiology of disease · 2014Article
- Fragile X mental retardation protein regulates trans-synaptic signaling in Drosophila.Disease models & mechanisms · 2013Article
- Programmed cell death is impaired in the developing brain of FMR1 mutants.Developmental neuroscience · 2013Article
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2 authors at 1 institution in 1 country.
Funding
Abstract
Fragile X syndrome (FXS), caused by loss of fragile X mental retardation 1 (FMR1) gene function, is the most common heritable cause of intellectual disability and autism spectrum disorders. The FMR1 product (FMRP) is an RNA-binding protein best established to function in activity-dependent modulation of synaptic connections. In the Drosophila FXS disease model, loss of functionally-conserved dFMRP causes synaptic overgrowth and overelaboration in pigment dispersing factor (PDF) peptidergic neurons in the adult brain. Here, we identify a very different component of PDF neuron misregulation in dfmr1 mutants: the aberrant retention of normally developmentally-transient PDF tritocerebral (PDF-TRI) neurons. In wild-type animals, PDF-TRI neurons in the central brain undergo programmed cell death and complete, processive clearance within days of eclosion. In the absence of dFMRP, a defective apoptotic program leads to constitutive maintenance of these peptidergic neurons. We tested whether this apoptotic defect is circuit-specific by examining crustacean cardioactive peptide (CCAP) and bursicon circuits, which are similarly developmentally-transient and normally eliminated immediately post-eclosion. In dfmr1 null mutants, CCAP/bursicon neurons also exhibit significantly delayed clearance dynamics, but are subsequently eliminated from the nervous system, in contrast to the fully persistent PDF-TRI neurons. Thus, the requirement of dFMRP for the retention of transitory peptidergic neurons shows evident circuit specificity. The novel defect of impaired apoptosis and aberrant neuron persistence in the Drosophila FXS model suggests an entirely new level of "pruning" dysfunction may contribute to the FXS disease state.
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