Evidence map›Paper›PMID 27940157›Full record

ArticleDevelopmental biology2017

Pa2G4 is a novel Six1 co-factor that is required for neural crest and otic development.

Karen M Neilson, Genevieve Abbruzzesse, Kristy Kenyon, Vanessa Bartolo, Patrick Krohn, Dominique Alfandari, Sally A Moody

Abstract read
In one paragraph

Article in Developmental biology, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 20 papers.

0numbers the graph read from it
0cells of the map it votes in
20citing papers in PubMed
1.6field-weighted citation impact, top 16% 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

20 citing papers in PubMed, 29 citations in OpenAlex.

  1. Review
  2. Article
  3. Using Xenopus to discover new candidate genes involved in BOR and other congenital hearing loss syndromes.Journal of experimental zoology. Part B, Molecular and developmental evolution · 2024
    Review
  4. Article
  5. The sulfotransferase XB5850668.L is required to apportion embryonic ectodermal domains.Developmental dynamics : an official publication of the American Association of Anatomists · 2023
    Article
  6. Article
  7. Ablation of ZC3H11A causes early embryonic lethality and dysregulation of metabolic processes.Proceedings of the National Academy of Sciences of the United States of America · 2023
    Article
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  17. Roles of ErbB3-binding protein 1 (EBP1) in embryonic development and gene-silencing control.Proceedings of the National Academy of Sciences of the United States of America · 2019
    Article
  18. Article
  19. Article
  20. Xenbase: Facilitating the Use ofFrontiers in physiology · 2019
    Review
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

7 authors at 3 institutions in 1 country.

Karen M NeilsonDepartment of Anatomy and Regenerative Biology, George Washington University, School of Medicine and Health Sciences, Washington, DC, USA.
Genevieve AbbruzzesseDepartment of Veterinary and Animal Sciences, University of Massachusetts Amherst, Amherst, MA, USA.
Kristy KenyonDepartment of Biology, Hobart and William Smith Colleges, Geneva, NY, USA.
Vanessa BartoloDepartment of Veterinary and Animal Sciences, University of Massachusetts Amherst, Amherst, MA, USA.
Patrick KrohnDepartment of Anatomy and Regenerative Biology, George Washington University, School of Medicine and Health Sciences, Washington, DC, USA.
Dominique AlfandariDepartment of Veterinary and Animal Sciences, University of Massachusetts Amherst, Amherst, MA, USA.
Sally A MoodyDepartment of Anatomy and Regenerative Biology, George Washington University, School of Medicine and Health Sciences, Washington, DC, USA. Electronic address: samoody@gwu.edu.
George Washington University · USUniversity of Massachusetts Amherst · USHobart and William Smith Colleges · US

Funding

Research_ProjectU54HD090257 · NICHD · CHILDREN'S RESEARCH INSTITUTE · PI GUAY-WOODFORD, LISA MARIE · 2016 to 2020
$5.9M
Mechanism of Xenopus Cranial Neural Crest Cell MigrationR01DE016289 · NIDCR · UNIVERSITY OF MASSACHUSETTS AMHERST · PI ALFANDARI, DOMINIQUE R · 2006 to 2022
$5.4M
Gene Regulation of Cranial Sensory Placode DevelopmentR01DE022065 · NIDCR · GEORGE WASHINGTON UNIVERSITY · PI MOODY, SALLY ANN · 2011 to 2015
$1.7M
Novel Six1 co-factors and their role in placode developmentR03HD055321 · NICHD · GEORGE WASHINGTON UNIVERSITY · PI NEILSON, KAREN MARY · 2009 to 2010
$156k
NICHD NIH HHS R03 HD055321NICHD NIH HHS U54 HD090257NIDCR NIH HHS R01 DE016289NIDCR NIH HHS R01 DE022065
6 · The paper itself

Abstract

Mutations in SIX1 and in its co-factor, EYA1, underlie Branchiootorenal Spectrum disorder (BOS), which is characterized by variable branchial arch, otic and kidney malformations. However, mutations in these two genes are identified in only half of patients. We screened for other potential co-factors, and herein characterize one of them, Pa2G4 (aka Ebp1/Plfap). In human embryonic kidney cells, Pa2G4 binds to Six1 and interferes with the Six1-Eya1 complex. In Xenopus embryos, knock-down of Pa2G4 leads to down-regulation of neural border zone, neural crest and cranial placode genes, and concomitant expansion of neural plate genes. Gain-of-function leads to a broader neural border zone, expanded neural crest and altered cranial placode domains. In loss-of-function assays, the later developing otocyst is reduced in size, which impacts gene expression. In contrast, the size of the otocyst in gain-of-function assays is not changed but the expression domains of several otocyst genes are reduced. Together these findings establish an interaction between Pa2G4 and Six1, and demonstrate that it has an important role in the development of tissues affected in BOS. Thereby, we suggest that pa2g4 is a potential candidate gene for BOS.

Indexed as

Amino Acid SequenceAnimalsCell DeathCell ProliferationEar, InnerEmbryo, NonmammalianFaceGene Expression Regulation, DevelopmentalGene Knockdown TechniquesHEK293 CellsHomeodomain ProteinsHumansNeural CrestNeural PlateProtein BindingProtein DomainsHomeodomain ProteinsPA2G4 protein, XenopusSix1 protein, XenopusXenopus ProteinsBranchiootorenalEbp1Eya1PlfapSIX1

Identifiers

PMID27940157
PMCPMC5221411
OpenAlexW2560004660

What OpenQuestion holds

Textmetadata
LicenceTDM
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.