Evidence map›Paper›PMID 34002468›Full record

ArticleGenes, brain, and behavior2022

Profiling beneficial and potential adverse effects of MeCP2 overexpression in a hypomorphic Rett syndrome mouse model.

Sheryl Anne D Vermudez, Rocco G Gogliotti, Bright Arthur, Aditi Buch, Clarissa Morales, Yuta Moxley, Hemangi Rajpal, P Jeffrey Conn, Colleen M Niswender

Open access · hybridAbstract read
In one paragraph

Article in Genes, brain, and behavior, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 13 papers.

0numbers the graph read from it
0cells of the map it votes in
13citing papers in PubMed
2.1field-weighted citation impact, top 13% of its field
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

13 citing papers in PubMed, 12 citations in OpenAlex.

  1. Review
  2. Article
  3. Article
  4. MeCP2 gene therapy ameliorates disease phenotype in mouse model for Pitt Hopkins syndrome.Neurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics · 2024
    Article
  5. Review
  6. Inhibitory Synaptic Influences on Developmental Motor Disorders.International journal of molecular sciences · 2023
    Review
  7. State-of-the-art therapies for Rett syndrome.Developmental medicine and child neurology · 2023
    Review
  8. Review
  9. Article
  10. Article
  11. Gene therapy for Rett syndrome.Genes, brain, and behavior · 2022
    Article
  12. Article
  13. Review
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

9 authors at 2 institutions in 1 country.

Sheryl Anne D VermudezDepartment of Pharmacology and Warren Center for Neuroscience Drug Discovery, Vanderbilt University, Nashville, Tennessee, USA.ORCID 0000-0002-7020-8347
Rocco G GogliottiDepartment of Molecular Pharmacology and Neuroscience, Loyola University Chicago, Chicago, Illinois, USA.
Bright ArthurDepartment of Pharmacology and Warren Center for Neuroscience Drug Discovery, Vanderbilt University, Nashville, Tennessee, USA.
Aditi BuchDepartment of Pharmacology and Warren Center for Neuroscience Drug Discovery, Vanderbilt University, Nashville, Tennessee, USA.
Clarissa MoralesDepartment of Pharmacology and Warren Center for Neuroscience Drug Discovery, Vanderbilt University, Nashville, Tennessee, USA.
Yuta MoxleyDepartment of Pharmacology and Warren Center for Neuroscience Drug Discovery, Vanderbilt University, Nashville, Tennessee, USA.
Hemangi RajpalDepartment of Pharmacology and Warren Center for Neuroscience Drug Discovery, Vanderbilt University, Nashville, Tennessee, USA.
P Jeffrey ConnDepartment of Pharmacology and Warren Center for Neuroscience Drug Discovery, Vanderbilt University, Nashville, Tennessee, USA.
Colleen M NiswenderDepartment of Pharmacology and Warren Center for Neuroscience Drug Discovery, Vanderbilt University, Nashville, Tennessee, USA.
Vanderbilt University · USLoyola University Chicago · US

Funding

TRAINING IN PHARMACOLOGICAL SCIENCEST32GM007628 · NIGMS · VANDERBILT UNIVERSITY · PI BARNETT, JOEY VICTOR, KAVALALI, EGE T · 1985 to 2022
$11.2M
Overall: Eunice Kennedy Shriver Intellectual and Developmental Disabilities Research Center at VanderbiltP50HD103537 · NICHD · VANDERBILT UNIVERSITY MEDICAL CENTER · PI Lea K Davis · 2020 to 2026
$10.3M
A precision medicine approach to Rett SyndromeR01NS112171 · NINDS · LOYOLA UNIVERSITY CHICAGO · PI GOGLIOTTI, ROCCO GEORGE · 2020 to 2024
$1.7M
Normalizing E:I imbalance in Rett Syndrome by Modulation of Late Response GenesK01MH112983 · NIMH · VANDERBILT UNIVERSITY · PI GOGLIOTTI, ROCCO GEORGE · 2017 to 2020
$543k
Stratifying Rett syndrome patient populations to evaluate the efficacy of genetic and pharmacological therapeutic interventionsF31MH119699 · NIMH · VANDERBILT UNIVERSITY · PI VERMUDEZ, SHERYL ANNE DAGUIMOL · 2019 to 2021
$91k
NICHD NIH HHS P50 HD103537NIGMS NIH HHS T32 GM007628NIMH NIH HHS F31 MH119699NIMH NIH HHS K01 MH112983NINDS NIH HHS R01 NS112171
6 · The paper itself

Abstract

De novo loss-of-function mutations in methyl-CpG-binding protein 2 (MeCP2) lead to the neurodevelopmental disorder Rett syndrome (RTT). Despite promising results from strategies aimed at increasing MeCP2 levels, additional studies exploring how hypomorphic MeCP2 mutations impact the therapeutic window are needed. Here, we investigated the consequences of genetically introducing a wild-type MECP2 transgene in the Mecp2 R133C mouse model of RTT. The MECP2 transgene reversed the majority of RTT-like phenotypes exhibited by male and female Mecp2 R133C mice. However, three core symptom domains were adversely affected in female Mecp2

Indexed as

PhenotypeAnimalsFemaleGenetic TherapyMaleMethyl-CpG-Binding Protein 2MiceMice, Inbred C57BLMutationRett SyndromeMecp2 protein, mouseMethyl-CpG-Binding Protein 2MeCP2MECP2 duplication syndromemouse modelneurodevelopmentRett syndrome

Identifiers

PMID34002468
PMCPMC8599502
OpenAlexW3162474665

What OpenQuestion 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 OpenQuestion graph.