ArticleNeuron2023
Species-specific FMRP regulation of RACK1 is critical for prenatal cortical development.
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Article in Neuron, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 22 papers.
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22citing papers in PubMed
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Who cites it
22 citing papers in PubMed, 33 citations in OpenAlex.
- The mMolecular psychiatry · 2026Article
- KIF1A-mediated trafficking is required for neuronal autophagy in human neurons.bioRxiv : the preprint server for biology · 2026Article
- RACK1 governs a dual metabolic switch in lung adenocarcinoma through c-Src/G6PD and TRIM21/LDHA Axes.Cell death & disease · 2026Article
- Mitochondrial dynamics in neurodevelopment and neurodevelopmental disorders.Nature reviews. Neuroscience · 2026Review
- Suppression of astrocyte BMP signaling improves molecular signatures and functional deficits in a fragile X syndrome mouse model.Nature communications · 2026Article
- A semi-automated MEA spike sorting method for high-throughput assessment of cultured neurons.Stem cell reports · 2026Article
- Article
- Metabolic reprogramming during human neuron differentiation indicates glutaminase as a key determinant in Fragile X syndrome.Cell reports · 2026Article
- FMRP regulation of STAT3-MYC signaling is critical for adult hippocampal neurogenesis and cognitive flexibility.Cell death and differentiation · 2026Article
- The role of mitochondria-endoplasmic reticulum crosstalk in colorectal cancer.Genes & diseases · 2026Review
- An Emerging Role for OGDHL: From Mitochondrial Energy Metabolism to Neurodevelopmental Disorders.Biology · 2025Review
- A Duality of Function: An Integrative Model of RACK1 as a Switch Between Translational and Signaling Hubs.International journal of molecular sciences · 2025Review
- Sleep in a mouse model of fragile X syndrome is resistant to metabolic manipulations.Human molecular genetics · 2025Article
- Multimodal analyses reveal genes driving electrophysiological maturation of neurons in the primate prefrontal cortex.Neuron · 2025Article
- The 40S ribosomal subunit recycling complex modulates mitochondrial dynamics and endoplasmic reticulum - mitochondria tethering at mitochondrial fission/fusion hotspots.Nature communications · 2025Article
- Consequences of trisomy 21 for brain development in Down syndrome.Nature reviews. Neuroscience · 2024Review
- Advances in human pluripotent stem cell reporter systems.iScience · 2024Review
- Dysregulated Purinergic Signalling in Fragile X Syndrome Cortical Astrocytes.Neuromolecular medicine · 2024Article
- CGG repeats in the human FMR1 gene regulate mRNA localization and cellular stress in developing neurons.Cell reports · 2024Article
- From wings to whiskers to stem cells: why every model matters in fragile X syndrome research.Journal of neurodevelopmental disorders · 2024Review
Corrections and comments
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Authors and funding
26 authors at 2 institutions in 1 country.
Minjie ShenWaisman Center, University of Wisconsin-Madison, Madison, WI 53705, USA; Department of Neuroscience, School of Medicine and Public Health, University of Wisconsin-Madison, Madison, WI 53705, USA.
Carissa L SiroisWaisman Center, University of Wisconsin-Madison, Madison, WI 53705, USA; Department of Neuroscience, School of Medicine and Public Health, University of Wisconsin-Madison, Madison, WI 53705, USA.
Yu GuoWaisman Center, University of Wisconsin-Madison, Madison, WI 53705, USA; Department of Neuroscience, School of Medicine and Public Health, University of Wisconsin-Madison, Madison, WI 53705, USA.
Meng LiWaisman Center, University of Wisconsin-Madison, Madison, WI 53705, USA; Department of Neuroscience, School of Medicine and Public Health, University of Wisconsin-Madison, Madison, WI 53705, USA.
Qiping DongWaisman Center, University of Wisconsin-Madison, Madison, WI 53705, USA.
Natasha M Méndez-AlbeloWaisman Center, University of Wisconsin-Madison, Madison, WI 53705, USA; Department of Neuroscience, School of Medicine and Public Health, University of Wisconsin-Madison, Madison, WI 53705, USA; Molecular Cellular Pharmacology Training Program, University of Wisconsin-Madison, Madison, WI 53705, USA.
Yu GaoWaisman Center, University of Wisconsin-Madison, Madison, WI 53705, USA; Department of Neuroscience, School of Medicine and Public Health, University of Wisconsin-Madison, Madison, WI 53705, USA.
Saniya KhullarWaisman Center, University of Wisconsin-Madison, Madison, WI 53705, USA; Departments of Biostatistics and Medical Informatics, School of Medicine and Public Health, University of Wisconsin-Madison, Madison, WI 53705, USA.
Lee KisselNeuroscience Training Program, University of Wisconsin-Madison, Madison, WI 53705, USA.
Soraya O SandovalWaisman Center, University of Wisconsin-Madison, Madison, WI 53705, USA; Department of Neuroscience, School of Medicine and Public Health, University of Wisconsin-Madison, Madison, WI 53705, USA; Neuroscience Training Program, University of Wisconsin-Madison, Madison, WI 53705, USA.
Natalie E WolkoffWaisman Center, University of Wisconsin-Madison, Madison, WI 53705, USA; Department of Neuroscience, School of Medicine and Public Health, University of Wisconsin-Madison, Madison, WI 53705, USA.
Sabrina X HuangWaisman Center, University of Wisconsin-Madison, Madison, WI 53705, USA; Department of Neuroscience, School of Medicine and Public Health, University of Wisconsin-Madison, Madison, WI 53705, USA.
Zhiyan XuWaisman Center, University of Wisconsin-Madison, Madison, WI 53705, USA; Department of Neuroscience, School of Medicine and Public Health, University of Wisconsin-Madison, Madison, WI 53705, USA; Graduate Program in Cell and Molecular Biology, University of Wisconsin-Madison, Madison, WI 53705, USA.
Jonathan E BryanWaisman Center, University of Wisconsin-Madison, Madison, WI 53705, USA; Departments of Biostatistics and Medical Informatics, School of Medicine and Public Health, University of Wisconsin-Madison, Madison, WI 53705, USA.
Amaya M ContractorWaisman Center, University of Wisconsin-Madison, Madison, WI 53705, USA; Department of Neuroscience, School of Medicine and Public Health, University of Wisconsin-Madison, Madison, WI 53705, USA.
Tomer KorabelnikovWaisman Center, University of Wisconsin-Madison, Madison, WI 53705, USA; Department of Neuroscience, School of Medicine and Public Health, University of Wisconsin-Madison, Madison, WI 53705, USA.
Ian A GlassBirth Defects Research Laboratory, University of Washington, Seattle, WA 98195, USA.
Dan DohertyBirth Defects Research Laboratory, University of Washington, Seattle, WA 98195, USA.
Birth Defects Research LaboratoryBirth Defects Research Laboratory, University of Washington, Seattle, WA 98195, USA.
Jon E LevineDepartment of Neuroscience, School of Medicine and Public Health, University of Wisconsin-Madison, Madison, WI 53705, USA; Wisconsin National Primate Research Center, University of Wisconsin-Madison, Madison, WI 53715, USA.
André M M SousaWaisman Center, University of Wisconsin-Madison, Madison, WI 53705, USA; Department of Neuroscience, School of Medicine and Public Health, University of Wisconsin-Madison, Madison, WI 53705, USA.
Qiang ChangWaisman Center, University of Wisconsin-Madison, Madison, WI 53705, USA; Department of Neurology, School of Medicine and Public Health, University of Wisconsin-Madison, Madison, WI 53705, USA; Laboratory of Genetics, University of Wisconsin-Madison, Madison, WI 53706, USA.
Anita BhattacharyyaWaisman Center, University of Wisconsin-Madison, Madison, WI 53705, USA; Department of Cell and Regenerative Biology, School of Medicine and Public Health, University of Wisconsin-Madison, Madison, WI 53705, USA.
Daifeng WangWaisman Center, University of Wisconsin-Madison, Madison, WI 53705, USA; Departments of Biostatistics and Medical Informatics, School of Medicine and Public Health, University of Wisconsin-Madison, Madison, WI 53705, USA.
Donna M WerlingLaboratory of Genetics, University of Wisconsin-Madison, Madison, WI 53706, USA.
Xinyu ZhaoWaisman Center, University of Wisconsin-Madison, Madison, WI 53705, USA; Department of Neuroscience, School of Medicine and Public Health, University of Wisconsin-Madison, Madison, WI 53705, USA. Electronic address: xinyu.zhao@wisc.edu.
University of Wisconsin–Madison · USUniversity of Washington · US
Funding
WNPRC Supplemental Request for Nonhuman Primate Enclosures to Equip HIV/AIDS-Related Research FacilitiesP51OD011106 · OD · UNIVERSITY OF WISCONSIN-MADISON · PI Dorota A. Grejner-Brzezinska · 2012 to 2026
$150.7MLABORATORY OF DEVELOPMENTAL BIOLOGYR24HD000836 · NICHD · UNIVERSITY OF WASHINGTON · PI Ian Amos Glass · 1995 to 2026
$16.9MUnderstanding the molecular mechanisms that contribute to neuropsychiatric symptoms in Alzheimer DiseaseR01AG067025 · NIA · ICAHN SCHOOL OF MEDICINE AT MOUNT SINAI · PI FINKBEINER, STEVEN M, HAROUTUNIAN, VAHRAM · 2019 to 2023
$11.8MWaisman Center Intellectual and Developmental Disabilities Research CenterP50HD105353 · NICHD · UNIVERSITY OF WISCONSIN-MADISON · PI Qiang Chang · 2021 to 2026
$8.5MDiscovery and validation of neuronal enhancers as development of psychiatric disorders supplementU01MH116492 · NIMH · UNIV OF MASSACHUSETTS MED SCH WORCESTER · PI GERSTEIN, MARK BENDER, WENG, ZHIPING · 2018 to 2023
$6.2MWaisman Intellectual and Developmental Disabilities Research Center Down Syndrome RegistryU54HD090256 · NICHD · UNIVERSITY OF WISCONSIN-MADISON · PI POLLAK, SETH D · 2016 to 2020
$6.0MNewborn Neurons in the Adult Hippocampal NetworkR01NS064025 · NINDS · UNIVERSITY OF ALABAMA AT BIRMINGHAM · PI OVERSTREET-WADICHE, LINDA · 2009 to 2023
$5.6MGraduate Training in Molecular and Cellular PharmacologyT32GM141013 · NIGMS · UNIVERSITY OF WISCONSIN-MADISON · PI Anjon Audhya, Aaron Matthew LeBeau · 2021 to 2026
$3.2MIn Vivo Function of Neuronal Activity-Induced MeCP2 phosphorylationR01HD064743 · NICHD · UNIVERSITY OF WISCONSIN-MADISON · PI CHANG, QIANG · 2010 to 2021
$3.0MThe Role of MDM2 in FMRP regulation of neuronal developmentR01MH116582 · NIMH · UNIVERSITY OF WISCONSIN-MADISON · PI ZHAO, XINYU · 2018 to 2022
$2.2MThe role of RNA binding protein in FXR1P in interneuronsR01MH118827 · NIMH · UNIVERSITY OF WISCONSIN-MADISON · PI ZHAO, XINYU · 2019 to 2023
$2.2MIntegration of Experience-Induced Gene Expression and Circuit FunctionsR01NS105200 · NINDS · UNIVERSITY OF WISCONSIN-MADISON · PI JACKSON, MEYER B., ZHAO, XINYU · 2018 to 2022
$2.0MNIA NIH HHS R01 AG067025NICHD NIH HHS P50 HD105353NICHD NIH HHS R01 HD064743NICHD NIH HHS R24 HD000836NICHD NIH HHS U54 HD090256NIGMS NIH HHS T32 GM141013NIH HHS P51 OD011106NIMH NIH HHS R01 MH116582NIMH NIH HHS R01 MH118827NIMH NIH HHS R36 MH136790NIMH NIH HHS U01 MH116492NINDS NIH HHS R01 NS064025NINDS NIH HHS R01 NS105200NINDS NIH HHS R21 NS105339
Abstract
Fragile X messenger ribonucleoprotein 1 protein (FMRP) deficiency leads to fragile X syndrome (FXS), an autism spectrum disorder. The role of FMRP in prenatal human brain development remains unclear. Here, we show that FMRP is important for human and macaque prenatal brain development. Both FMRP-deficient neurons in human fetal cortical slices and FXS patient stem cell-derived neurons exhibit mitochondrial dysfunctions and hyperexcitability. Using multiomics analyses, we have identified both FMRP-bound mRNAs and FMRP-interacting proteins in human neurons and unveiled a previously unknown role of FMRP in regulating essential genes during human prenatal development. We demonstrate that FMRP interaction with CNOT1 maintains the levels of receptor for activated C kinase 1 (RACK1), a species-specific FMRP target. Genetic reduction of RACK1 leads to both mitochondrial dysfunctions and hyperexcitability, resembling FXS neurons. Finally, enhancing mitochondrial functions rescues deficits of FMRP-deficient cortical neurons during prenatal development, demonstrating targeting mitochondrial dysfunction as a potential treatment.
Indexed as
Autism Spectrum DisorderFragile X SyndromeMitochondrial DiseasesFragile X Messenger Ribonucleoprotein 1HumansNeoplasm ProteinsNeurogenesisNeuronsReceptors for Activated C KinaseTranscription FactorsCNOT1 protein, humanFragile X Messenger Ribonucleoprotein 1Neoplasm ProteinsRACK1 protein, humanReceptors for Activated C KinaseTranscription Factorsex vivo cortical slicesFMR1FMRP interactorfragile X syndromehuman specifici physiologymacaquemitochondriapluripoent stem cellsRACK1
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