Evidence map›Paper›PMID 32066685›Full record

ArticleTranslational psychiatry2020

Translational outcomes in a full gene deletion of ubiquitin protein ligase E3A rat model of Angelman syndrome.

E L Berg, M C Pride, S P Petkova, R D Lee, N A Copping, Y Shen, A Adhikari, T A Fenton, L R Pedersen, L S Noakes and 18 more

Open access · goldAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Translational psychiatry, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 33 papers.

0numbers the graph read from it
0cells of the map it votes in
33citing papers in PubMed
10.1field-weighted citation impact, top 1% 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

33 citing papers in PubMed, 65 citations in OpenAlex.

  1. Article
  2. Article
  3. Article
  4. Review
  5. Article
  6. Article
  7. Review
  8. Article
  9. Article
  10. Article
  11. Stem cell models of Angelman syndrome.Frontiers in cell and developmental biology · 2023
    Review
  12. Article
  13. Article
  14. Article
  15. Improving Gene Therapy for Angelman Syndrome with Secreted Human UBE3A.Neurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics · 2022
    Article
  16. Review
  17. Gait as a quantitative translational outcome measure in Angelman syndrome.Autism research : official journal of the International Society for Autism Research · 2022
    Article
  18. Article
  19. Article
  20. Excessive Laughter-like Vocalizations, Microcephaly, and Translational Outcomes in theThe Journal of neuroscience : the official journal of the Society for Neuroscience · 2021
    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

28 authors at 9 institutions in 4 countries.

E L BergMIND Institute and Department of Psychiatry and Behavioral Sciences, University of California Davis School of Medicine, Sacramento, CA, USA.ORCID http://orcid.org/0000-0001-6413-2561
M C PrideMIND Institute and Department of Psychiatry and Behavioral Sciences, University of California Davis School of Medicine, Sacramento, CA, USA.
S P PetkovaMIND Institute and Department of Psychiatry and Behavioral Sciences, University of California Davis School of Medicine, Sacramento, CA, USA.
R D LeeMIND Institute and Department of Psychiatry and Behavioral Sciences, University of California Davis School of Medicine, Sacramento, CA, USA.
N A CoppingMIND Institute and Department of Psychiatry and Behavioral Sciences, University of California Davis School of Medicine, Sacramento, CA, USA.
Y ShenMIND Institute and Department of Psychiatry and Behavioral Sciences, University of California Davis School of Medicine, Sacramento, CA, USA.
A AdhikariMIND Institute and Department of Psychiatry and Behavioral Sciences, University of California Davis School of Medicine, Sacramento, CA, USA.
T A FentonMIND Institute and Department of Psychiatry and Behavioral Sciences, University of California Davis School of Medicine, Sacramento, CA, USA.
L R PedersenMIND Institute and Department of Psychiatry and Behavioral Sciences, University of California Davis School of Medicine, Sacramento, CA, USA.
L S NoakesMouse Imaging Centre, Toronto Centre for Phenogenomics, The Hospital for Sick Children, Toronto, ON, Canada.
B J NiemanMouse Imaging Centre, Toronto Centre for Phenogenomics, The Hospital for Sick Children, Toronto, ON, Canada.
J P LerchWellcome Centre for Integrative Neuroimaging, The University of Oxford, Oxford, UK.
S HarrisDepartment of Pediatrics and Neurology, Baylor College of Medicine, Houston, TX, USA.
H A BornDepartment of Pediatrics and Neurology, Baylor College of Medicine, Houston, TX, USA.ORCID http://orcid.org/0000-0001-9934-648X
M M PetersDepartment of Molecular Pharmacology and Physiology, University of South Florida, Tampa, FL, USA.
P DengStem Cell Program, Institute for Regenerative Cures, and Department of Neurology, University of California Davis School of Medicine, Sacramento, CA, USA.
D L CameronStem Cell Program, Institute for Regenerative Cures, and Department of Neurology, University of California Davis School of Medicine, Sacramento, CA, USA.
K D FinkStem Cell Program, Institute for Regenerative Cures, and Department of Neurology, University of California Davis School of Medicine, Sacramento, CA, USA.
U BeitnereMIND Institute, Genome Center, and Department of Biochemistry and Molecular Medicine, University of California Davis, Davis, CA, USA.
H O'GeenMIND Institute, Genome Center, and Department of Biochemistry and Molecular Medicine, University of California Davis, Davis, CA, USA.
A E AndersonDepartment of Pediatrics and Neurology, Baylor College of Medicine, Houston, TX, USA.
S V DindotDepartment of Veterinary Pathobiology, College of Veterinary Medicine and Biomedical Sciences, Texas A&M University, College Station, TX, USA.
K R NashDepartment of Molecular Pharmacology and Physiology, University of South Florida, Tampa, FL, USA.
E J WeeberDepartment of Molecular Pharmacology and Physiology, University of South Florida, Tampa, FL, USA.
M WöhrBehavioral Neuroscience, Experimental and Biological Psychology, Philipps-University of Marburg, Marburg, Germany.ORCID http://orcid.org/0000-0001-6986-5684
J EllegoodMouse Imaging Centre, Toronto Centre for Phenogenomics, The Hospital for Sick Children, Toronto, ON, Canada.ORCID http://orcid.org/0000-0003-1504-3321
D J SegalMIND Institute, Genome Center, and Department of Biochemistry and Molecular Medicine, University of California Davis, Davis, CA, USA.ORCID http://orcid.org/0000-0001-8962-3105
J L SilvermanMIND Institute and Department of Psychiatry and Behavioral Sciences, University of California Davis School of Medicine, Sacramento, CA, USA. jsilverman@ucdavis.edu.ORCID http://orcid.org/0000-0001-9357-5476
University of California, Davis · USBaylor College of Medicine · USCalifornia Institute for Regenerative Medicine · USHospital for Sick Children · CAUniversity of South Florida · USPhilipps University of Marburg · DETexas A&M University · USUniversity of California System · USUniversity of Oxford · GB

Funding

Research Project: Pathologic Significance of Maternal AutoantibodiesP50HD103526 · NICHD · UNIVERSITY OF CALIFORNIA AT DAVIS · PI LEONARD J. ABBEDUTO, Melissa Dawn Bauman · 2020 to 2026
$9.7M
Rodent Behavior CoreU54HD079125 · NICHD · UNIVERSITY OF CALIFORNIA AT DAVIS · PI VAN DE WATER, JUDY A. · 2013 to 2019
$8.5M
Phenotypic Characterization of Novel Models of Dup15q SyndromeR01NS097808 · NINDS · UNIVERSITY OF CALIFORNIA AT DAVIS · PI SILVERMAN, JILL LYNN · 2017 to 2021
$1.7M
NICHD NIH HHS P50 HD103526NICHD NIH HHS U54 HD079125NINDS NIH HHS R01 NS097808
6 · The paper itself

Abstract

Angelman syndrome (AS) is a rare neurodevelopmental disorder characterized by developmental delay, impaired communication, motor deficits and ataxia, intellectual disabilities, microcephaly, and seizures. The genetic cause of AS is the loss of expression of UBE3A (ubiquitin protein ligase E6-AP) in the brain, typically due to a deletion of the maternal 15q11-q13 region. Previous studies have been performed using a mouse model with a deletion of a single exon of Ube3a. Since three splice variants of Ube3a exist, this has led to a lack of consistent reports and the theory that perhaps not all mouse studies were assessing the effects of an absence of all functional UBE3A. Herein, we report the generation and functional characterization of a novel model of Angelman syndrome by deleting the entire Ube3a gene in the rat. We validated that this resulted in the first comprehensive gene deletion rodent model. Ultrasonic vocalizations from newborn Ube3a

Indexed as

Angelman SyndromeIntellectual DisabilityAnimalsGene DeletionMemoryRatsUbiquitin-Protein LigasesUbe3a protein, ratUbiquitin-Protein Ligases

Identifiers

PMID32066685
PMCPMC7026078
OpenAlexW3001089571

What OpenQuestion holds

Textmetadata
LicenceCC BY
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.