Evidence map›Paper›PMID 20150232›Full record

ArticleHuman molecular genetics2010

AP-2alpha knockout mice exhibit optic cup patterning defects and failure of optic stalk morphogenesis.

Erin A Bassett, Trevor Williams, Amanda L Zacharias, Philip J Gage, Sabine Fuhrmann, Judith A West-Mays

Open access · bronzeAbstract read
In one paragraph

Article in Human molecular genetics, 2010. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 43 papers.

0numbers the graph read from it
0cells of the map it votes in
43citing papers in PubMed
2.4field-weighted citation impact, top 11% 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

43 citing papers in PubMed, 69 citations in OpenAlex.

  1. Article
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  7. Pax2a, but not pax2b, influences cell survival and periocular mesenchyme localization to facilitate zebrafish optic fissure closure.Developmental dynamics : an official publication of the American Association of Anatomists · 2022
    Article
  8. Article
  9. Porcn is essential for growth and invagination of the mammalian optic cup.Frontiers in cell and developmental biology · 2022
    Article
  10. Eyes on CHARGE syndrome: Roles of CHD7 in ocular development.Frontiers in cell and developmental biology · 2022
    Review
  11. Article
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  14. Article
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  16. A Heterozygous Novel Mutation inFrontiers in pediatrics · 2020
    Article
  17. Review
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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

6 authors at 4 institutions in 2 countries.

Erin A BassettDepartment of Pathology and Molecular Medicine, McMaster University, Hamilton, ON, Canada.
Trevor Williams
Amanda L Zacharias
Philip J Gage
Sabine Fuhrmann
Judith A West-Mays
McMaster University · CAUniversity of Colorado Anschutz Medical Campus · USUniversity of Michigan · USUniversity of Utah · US

Funding

VIVARIUM MODULEP30EY007003 · NEI · UNIVERSITY OF MICHIGAN AT ANN ARBOR · PI David Antonetti · 1987 to 2026
$17.6M
University of Utah, Core Vision Research GrantP30EY014800 · NEI · UTAH STATE HIGHER EDUCATION SYSTEM--UNIVERSITY OF UTAH · PI Jun Yang · 2005 to 2026
$14.6M
THE ROLE OF AP-2 IN CRANIOFACIAL DEVELOPMENTR01DE012728 · NIDCR · YALE UNIVERSITY · PI WILLIAMS, TREVOR J · 1998 to 2013
$5.5M
Role of Wnt/Frizzled signaling during eye developmentR01EY014954 · NEI · UNIVERSITY OF UTAH · PI FUHRMANN, SABINE · 2003 to 2013
$3.4M
Role of AP-2 genes in development of the lensR01EY011910 · NEI · TUFTS MEDICAL CENTER · PI WEST-MAYS, JUDITH A · 1998 to 2006
$1.3M
E75 IN THE DEVELOPING DROSOPHILA EYEF31EY007003 · NEI · YALE UNIVERSITY · PI SHILTON, ALYCIA N · 1999 to 2001
$14k
GENESIS OF NEURAL SPECIFICITY IN THE VISUAL SYSTEMR01EY001426 · NEI · NEW YORK MEDICAL COLLEGE · PI SHARMA, SANSAR C · 1985 to 1988
–
NEI NIH HHS EY007003NEI NIH HHS EY01426NEI NIH HHS EY014800NEI NIH HHS EY014954NEI NIH HHS EY11910NEI NIH HHS F31 EY007003NEI NIH HHS P30 EY007003NEI NIH HHS P30 EY014800NEI NIH HHS R01 EY011910NEI NIH HHS R01 EY014954NIDCR NIH HHS DE-12728NIDCR NIH HHS R01 DE012728
6 · The paper itself

Abstract

Appropriate development of the retina and optic nerve requires that the forebrain-derived optic neuroepithelium undergoes a precisely coordinated sequence of patterning and morphogenetic events, processes which are highly influenced by signals from adjacent tissues. Our previous work has suggested that transcription factor activating protein-2 alpha (AP-2alpha; Tcfap2a) has a non-cell autonomous role in optic cup (OC) development; however, it remained unclear how OC abnormalities in AP-2alpha knockout (KO) mice arise at the morphological and molecular level. In this study, we show that patterning and morphogenetic defects in the AP-2alpha KO optic neuroepithelium begin at the optic vesicle stage. During subsequent OC formation, ectopic neural retina and optic stalk-like tissue replaced regions of retinal pigment epithelium. AP-2alpha KO eyes also displayed coloboma in the ventral retina, and a rare phenotype in which the optic stalk completely failed to extend, causing the OCs to be drawn inward to the midline. We detected evidence of increased sonic hedgehog signaling in the AP-2alpha KO forebrain neuroepithelium, which likely contributed to multiple aspects of the ocular phenotype, including expansion of PAX2-positive optic stalk-like tissue into the OC. Our data suggest that loss of AP-2alpha in multiple tissues in the craniofacial region leads to severe OC and optic stalk abnormalities by disturbing the tissue-tissue interactions required for ocular development. In view of recent data showing that mutations in human TFAP2A result in similar eye defects, the current findings demonstrate that AP-2alpha KO mice provide a valuable model for human ocular disease.

Indexed as

Disease Models, AnimalAnimalsDNA PrimersEye AbnormalitiesFluorescent Antibody TechniqueGene Expression Regulation, DevelopmentalHedgehog ProteinsIn Situ HybridizationIn Situ Nick-End LabelingMiceMice, KnockoutMorphogenesisOptic NervePolymerase Chain ReactionProsencephalonRetinaDNA PrimersHedgehog ProteinsShh protein, mouseTranscription Factor AP-2

Identifiers

PMID20150232
PMCPMC2850623
OpenAlexW2114240336

What Socratic holds

Textmetadata
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.