Evidence map›Paper›PMID 33076843›Full record

ArticleJournal of neurodevelopmental disorders2020

Deficits in higher visual area representations in a mouse model of Angelman syndrome.

Leah B Townsend, Kelly A Jones, Christopher R Dorsett, Benjamin D Philpot, Spencer L Smith

Abstract read
In one paragraph

Article in Journal of neurodevelopmental disorders, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing 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

3 citing papers in PubMed.

  1. Article
  2. Article
  3. 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

5 authors.

Leah B TownsendNeuroscience Curriculum, University of North Carolina School of Medicine, Chapel Hill, NC, 27599, USA.
Kelly A JonesNeuroscience Curriculum, University of North Carolina School of Medicine, Chapel Hill, NC, 27599, USA.
Christopher R DorsettNeuroscience Curriculum, University of North Carolina School of Medicine, Chapel Hill, NC, 27599, USA.
Benjamin D PhilpotNeuroscience Curriculum, University of North Carolina School of Medicine, Chapel Hill, NC, 27599, USA.
Spencer L SmithDepartment of Electrical & Computer Engineering, Neuroscience Research Institute, Center for BioEngineering, University of California Santa Barbara, 2002 BioEngineering Building; Mail code 5100, Santa Barbara, CA, 93106-5100, USA. sls@ucsb.edu.ORCID 0000-0002-2021-7034

Funding

UNC Neuroscience Center Research Cores: MicroscopyP30NS045892 · NINDS · UNIV OF NORTH CAROLINA CHAPEL HILL · PI ZYLKA, MARK J. · 2003 to 2022
$11.5M
RESEARCH TRAINING IN THE NEUROSCIENCEST32NS007431 · NINDS · UNIV OF NORTH CAROLINA CHAPEL HILL · PI Juan Song, Mark J. Zylka · 1997 to 2026
$10.0M
Postdoctoral Research in Neurodevelopmental DisordersT32HD040127 · NICHD · UNIV OF NORTH CAROLINA CHAPEL HILL · PI BENJAMIN D PHILPOT, Mark D Shen · 2001 to 2026
$8.5M
UBE3A gain-of-function and parent-of-origin influence on neurodevelopmental phenotypesR01MH120229 · NIMH · UNIV OF NORTH CAROLINA CHAPEL HILL · PI PHILPOT, BENJAMIN D, ZYLKA, MARK J. · 2019 to 2023
$3.4M
White Matter Pathology in Angelman Syndrome and Its Potential as an Outcome Measure in Clinical TrialsR01HD093771 · NICHD · UNIV OF NORTH CAROLINA CHAPEL HILL · PI HAZLETT, HEATHER CODY, PHILPOT, BENJAMIN D · 2018 to 2022
$3.0M
Modulation of dendritic spiking in vivoR01NS091335 · NINDS · UNIV OF NORTH CAROLINA CHAPEL HILL · PI BRENMAN, JAY, SMITH, SPENCER LAVERE · 2015 to 2020
$2.0M
Development and function of higher visual areas in miceR01EY024294 · NEI · UNIV OF NORTH CAROLINA CHAPEL HILL · PI SMITH, SPENCER LAVERE · 2015 to 2019
$1.7M
Stimulus-driven cortical circuit deficits in a mouse model of Angelman SyndromeF31HD084174 · NICHD · UNIV OF NORTH CAROLINA CHAPEL HILL · PI TOWNSEND, LEAH BISHOP · 2015 to 2015
$17k
Howard Hughes Medical InstituteNational Institute of Child Health and Human Development R01HD093771National Institute of Child Health and Human Development R01MH120229National Institute of Child Health and Human Development T32HD40127NEI NIH HHS 024294NEI NIH HHS R01 EY024294NICHD NIH HHS F31 HD084174NICHD NIH HHS R01 HD093771NICHD NIH HHS T32 HD040127NIMH NIH HHS R01 MH120229NINDS NIH HHS P30 NS045892NINDS NIH HHS P30NS045892NINDS NIH HHS R01 NS091335NINDS NIH HHS R01NS091335NINDS NIH HHS T32 NS007431NINDS NIH HHS T32NS007431
6 · The paper itself

Abstract

backgroundSensory processing deficits are common in individuals with neurodevelopmental disorders. One hypothesis is that deficits may be more detectable in downstream, "higher" sensory areas. A mouse model of Angelman syndrome (AS), which lacks expression of the maternally inherited Ube3a allele, has deficits in synaptic function and experience-dependent plasticity in the primary visual cortex. Thus, we hypothesized that AS model mice have deficits in visually driven neuronal responsiveness in downstream higher visual areas (HVAs).

methodsHere, we used intrinsic signal optical imaging and two-photon calcium imaging to map visually evoked neuronal activity in the primary visual cortex and HVAs in response to an array of stimuli.

resultsWe found a highly specific deficit in HVAs. Drifting gratings that changed speed caused a strong response in HVAs in wildtype mice, but this was not observed in littermate AS model mice. Further investigation with two-photon calcium imaging revealed the effect to be largely driven by aberrant responses of inhibitory interneurons, suggesting a cellular basis for higher level, stimulus-selective cortical dysfunction in AS.

conclusionAssaying downstream, or "higher" circuitry may provide a more sensitive measure for circuit dysfunction in mouse models of neurodevelopmental disorders.

trial registrationNot applicable.

Indexed as

Angelman SyndromeVisual CortexAnimalsDisease Models, AnimalMiceNeurons

Identifiers

PMID33076843
PMCPMC7574469

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.