ArticleThe Journal of neuroscience : the official journal of the Society for Neuroscience2013
The disruption of Celf6, a gene identified by translational profiling of serotonergic neurons, results in autism-related behaviors.
Article in The Journal of neuroscience : the official journal of the Society for Neuroscience, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 60 papers.
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Who cites it
60 citing papers in PubMed, 100 citations in OpenAlex.
- CELF family of RNA-binding proteins: roles in disease biology and potential for therapeutic intervention.Cell communication and signaling : CCS · 2026Review
- Heterozygous CELF4 variants in the N-term region crucial for the RNA-binding activity lead to neurodevelopmental disorder and obesity.European journal of human genetics : EJHG · 2025Article
- Global analysis of excitotoxicity-induced alterations in RNA structure and RNA-protein binding in neurons.iScience · 2025Article
- The gain-of-function UBE3AScientific reports · 2025Article
- Perinatal oxycodone exposure causes long-term sex-dependent changes in weight trajectory and sensory processing in adult mice.Psychopharmacology · 2022Article
- Advanced paternal age diversifies individual trajectories of vocalization patterns in neonatal mice.iScience · 2022Article
- Article
- Rare and de novo variants in 827 congenital diaphragmatic hernia probands implicate LONP1 as candidate risk gene.American journal of human genetics · 2021Article
- Massively Parallel Reporter Assays: Defining Functional Psychiatric Genetic Variants Across Biological Contexts.Biological psychiatry · 2021Review
- Evolution of the Neocortex Through RNA-Binding Proteins and Post-transcriptional Regulation.Frontiers in neuroscience · 2021Review
- Ontogenetic Oxycodone Exposure Affects Early Life Communicative Behaviors, Sensorimotor Reflexes, and Weight Trajectory in Mice.Frontiers in behavioral neuroscience · 2021Article
- CLIP and Massively Parallel Functional Analysis of CELF6 Reveal a Role in Destabilizing Synaptic Gene mRNAs through Interaction with 3' UTR Elements.Cell reports · 2020Article
- DNMT3A Haploinsufficiency Results in Behavioral Deficits and Global Epigenomic Dysregulation Shared across Neurodevelopmental Disorders.Cell reports · 2020Article
- Control of cortical synapse development and plasticity by MET receptor tyrosine kinase, a genetic risk factor for autism.Journal of neuroscience research · 2020Review
- Genetic aetiology of self-harm ideation and behaviour.Scientific reports · 2020Article
- Translational derepression of Elavl4 isoforms at their alternative 5' UTRs determines neuronal development.Nature communications · 2020Article
- Neurosteroids as Selective Inhibitors of Glycine Receptor Activity: Structure-Activity Relationship Study on Endogenous Androstanes and Androstenes.Frontiers in molecular neuroscience · 2020Article
- Using animal models to evaluate the functional consequences of anesthesia during early neurodevelopment.Neurobiology of learning and memory · 2019Review
- Gtf2i and Gtf2ird1 mutation do not account for the full phenotypic effect of the Williams syndrome critical region in mouse models.Human molecular genetics · 2019Article
- RNA-binding protein CELF6 is cell cycle regulated and controls cancer cell proliferation by stabilizing p21.Cell death & disease · 2019Article
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Authors and funding
13 authors at 6 institutions in 2 countries.
Funding
Abstract
The immense molecular diversity of neurons challenges our ability to understand the genetic and cellular etiology of neuropsychiatric disorders. Leveraging knowledge from neurobiology may help parse the genetic complexity: identifying genes important for a circuit that mediates a particular symptom of a disease may help identify polymorphisms that contribute to risk for the disease as a whole. The serotonergic system has long been suspected in disorders that have symptoms of repetitive behaviors and resistance to change, including autism. We generated a bacTRAP mouse line to permit translational profiling of serotonergic neurons. From this, we identified several thousand serotonergic-cell expressed transcripts, of which 174 were highly enriched, including all known markers of these cells. Analysis of common variants near the corresponding genes in the AGRE collection implicated the RNA binding protein CELF6 in autism risk. Screening for rare variants in CELF6 identified an inherited premature stop codon in one of the probands. Subsequent disruption of Celf6 in mice resulted in animals exhibiting resistance to change and decreased ultrasonic vocalization as well as abnormal levels of serotonin in the brain. This work provides a reproducible and accurate method to profile serotonergic neurons under a variety of conditions and suggests a novel paradigm for gaining information on the etiology of psychiatric disorders.
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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.