Evidence map›Paper›PMID 38865241›Full record

ArticleCell reports2024

CGG repeats in the human FMR1 gene regulate mRNA localization and cellular stress in developing neurons.

Carissa L Sirois, Yu Guo, Meng Li, Natalie E Wolkoff, Tomer Korabelnikov, Soraya Sandoval, Jiyoun Lee, Minjie Shen, Amaya Contractor, Andre M M Sousa and 2 more

Abstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Cell reports, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.

0numbers the graph read from it
0cells of the map it votes in
6citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

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.

2 · The registry

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.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

6 citing papers in PubMed.

  1. Review
  2. Biomolecules · 2025
    Review
  3. Review
  4. Article
  5. Beyond the Synapse:International journal of molecular sciences · 2024
    Review
  6. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

12 authors.

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.
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.
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.
Soraya 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.
Jiyoun LeeWaisman 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.
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.
Amaya 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.
Andre 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.
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.
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.

Funding

Waisman Center Intellectual and Developmental Disabilities Research CenterP50HD105353 · NICHD · UNIVERSITY OF WISCONSIN-MADISON · PI Qiang Chang · 2021 to 2026
$8.5M
Waisman Intellectual and Developmental Disabilities Research Center Down Syndrome RegistryU54HD090256 · NICHD · UNIVERSITY OF WISCONSIN-MADISON · PI DAVIDSON, RICHARD J · 2016 to 2020
$6.0M
The Role of MDM2 in FMRP regulation of neuronal developmentR01MH116582 · NIMH · UNIVERSITY OF WISCONSIN-MADISON · PI ZHAO, XINYU · 2018 to 2022
$2.2M
The role of RNA binding protein in FXR1P in interneuronsR01MH118827 · NIMH · UNIVERSITY OF WISCONSIN-MADISON · PI ZHAO, XINYU · 2019 to 2023
$2.2M
Interrogate FMRP functions in primate brain developmentR01MH136152 · NIMH · UNIVERSITY OF WISCONSIN-MADISON · PI Xinyu Zhao · 2024 to 2026
$2.2M
Integration of Experience-Induced Gene Expression and Circuit FunctionsR01NS105200 · NINDS · UNIVERSITY OF WISCONSIN-MADISON · PI JACKSON, MEYER B., ZHAO, XINYU · 2018 to 2022
$2.0M
Identification of FMRP targets in human cortical neuronsR21NS105339 · NINDS · UNIVERSITY OF WISCONSIN-MADISON · PI BHATTACHARYYA, ANITA, ZHAO, XINYU · 2018 to 2019
$421k
Characterization of Fragile X Syndrome 3D Cortical OrganoidsR36MH136790 · NIMH · UNIVERSITY OF WISCONSIN-MADISON · PI SANDOVAL, SORAYA OROZCO · 2024 to 2025
$91k
NICHD NIH HHS P50 HD105353NICHD NIH HHS U54 HD090256NIMH NIH HHS R01 MH116582NIMH NIH HHS R01 MH118827NIMH NIH HHS R01 MH136152NIMH NIH HHS R36 MH136790NINDS NIH HHS R01 NS105200NINDS NIH HHS R21 NS105339
6 · The paper itself

Abstract

The human genome has many short tandem repeats, yet the normal functions of these repeats are unclear. The 5' untranslated region (UTR) of the fragile X messenger ribonucleoprotein 1 (FMR1) gene contains polymorphic CGG repeats, the length of which has differing effects on FMR1 expression and human health, including the neurodevelopmental disorder fragile X syndrome. We deleted the CGG repeats in the FMR1 gene (0CGG) in human stem cells and examined the effects on differentiated neurons. 0CGG neurons have altered subcellular localization of FMR1 mRNA and protein, and differential expression of cellular stress proteins compared with neurons with normal repeats (31CGG). In addition, 0CGG neurons have altered responses to glucocorticoid receptor (GR) activation, including FMR1 mRNA localization, GR chaperone HSP90α expression, GR localization, and cellular stress protein levels. Therefore, the CGG repeats in the FMR1 gene are important for the homeostatic responses of neurons to stress signals.

Indexed as

Fragile X Messenger Ribonucleoprotein 1NeuronsRNA, Messenger5' Untranslated RegionsHSP90 Heat-Shock ProteinsHumansReceptors, GlucocorticoidStress, PhysiologicalTrinucleotide Repeat ExpansionTrinucleotide Repeats5' Untranslated RegionsFMR1 protein, humanFragile X Messenger Ribonucleoprotein 1HSP90 Heat-Shock ProteinsReceptors, GlucocorticoidRNA, Messenger5′ UTRCGG repeatsCP: Molecular biologyCP: NeurosciencedexamethasoneFMR1FMRPfragile X syndromeglucocorticoid receptorHSP90human-specificneuronpluripotent stem cellsRNA localizationsingle molecule FISHstresstrinucleotide repeats

Identifiers

PMID38865241
PMCPMC11240841

What Socratic holds

Textmetadata
LicenceCC BY-NC-ND
Read underepoch 390

Registered trials

None linked

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 Socratic graph.