Evidence map›Paper›PMID 18483623›Full record

ArticleThe Journal of clinical investigation2008

Persistent expression of Pax3 in the neural crest causes cleft palate and defective osteogenesis in mice.

Meilin Wu, Jun Li, Kurt A Engleka, Bo Zhou, Min Min Lu, Joshua B Plotkin, Jonathan A Epstein

Open access · greenAbstract read
In one paragraph

Article in The Journal of clinical investigation, 2008. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 47 papers.

0numbers the graph read from it
0cells of the map it votes in
47citing papers in PubMed
2.9field-weighted citation impact, top 9% 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

47 citing papers in PubMed, 76 citations in OpenAlex.

  1. Review
  2. Article
  3. Article
  4. Genetic architecture of craniofacial morphogenesis: roles ofFrontiers in cell and developmental biology · 2026
    Review
  5. Association ofInternational journal of molecular sciences · 2025
    Article
  6. Article
  7. Article
  8. Article
  9. Article
  10. Article
  11. Journal of developmental biology · 2022
    Article
  12. Review
  13. Review
  14. Article
  15. Article
  16. Chemical-induced craniofacial anomalies caused by disruption of neural crest cell development in a zebrafish model.Developmental dynamics : an official publication of the American Association of Anatomists · 2020
    Article
  17. Article
  18. Article
  19. Article
  20. The True Story 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 2 institutions in 1 country.

Meilin WuDepartment of Cell and Developmental Biology and Penn Cardiovascular Institute, University of Pennsylvania, Philadelphia, Pennsylvania 19104, USA.
Jun Li
Kurt A Engleka
Bo Zhou
Min Min Lu
Joshua B Plotkin
Jonathan A Epstein
Cardiovascular Institute of the South · USUniversity of Pennsylvania · US

Funding

PAX3 REGULATION OF CARDIAC CONOTRUNCAL DEVELOPMENTR01HL061475 · NHLBI · UNIVERSITY OF PENNSYLVANIA · PI EPSTEIN, JONATHAN A. · 1999 to 2008
$3.2M
NHLBI NIH HHS R01 HL061475NHLBI NIH HHS R01 HL61475
6 · The paper itself

Abstract

Transcription factors regulate tissue patterning and cell fate determination during development; however, expression of early regulators frequently abates upon differentiation, suggesting that they may also play a role in maintaining an undifferentiated phenotype. The transcription factor paired box 3 (Pax3) is expressed by multipotent neural crest precursors and is implicated in neural crest disorders in humans such as Waardenburg syndrome. Pax3 is required for development of multiple neural crest lineages and for activation of lineage-specific programs, yet expression is generally extinguished once neural crest cells migrate from the dorsal neural tube and differentiate. Using a murine Cre-inducible system, we asked whether persistent Pax3 expression in neural crest derivatives would affect development or patterning. We found that persistent expression of Pax3 in cranial neural crest cells resulted in cleft palate, ocular defects, malformation of the sphenoid bone, and perinatal lethality. Furthermore, we demonstrated that Pax3 directly regulates expression of Sostdc1, a soluble inhibitor of bone morphogenetic protein (BMP) signaling. Persistent Pax3 expression renders the cranial crest resistant to BMP-induced osteogenesis. Thus, one mechanism by which Pax3 maintains the undifferentiated state of neural crest mesenchyme may be to block responsiveness to differentiation signals from the environment. These studies provide in vivo evidence for the importance of Pax3 downregulation during differentiation of multipotent neural crest precursors and cranial development.

Indexed as

Gene Expression Regulation, DevelopmentalOsteogenesisAnimalsBone DevelopmentCleft PalateDNA, ComplementaryDose-Response Relationship, DrugGenotypeMiceMice, TransgenicModels, BiologicalModels, GeneticNeural CrestPaired Box Transcription FactorsPAX3 Transcription FactorProteinsDNA, ComplementaryGt(ROSA)26Sor non-coding RNA, mousePaired Box Transcription FactorsPax3 protein, mousePAX3 Transcription FactorProteinsRNA, Untranslated

Identifiers

PMID18483623
PMCPMC2381747
OpenAlexW2022221183

What OpenQuestion 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 OpenQuestion graph.