Evidence map›Paper›PMID 28346501›Full record

ArticlePloS one2017

A tissue-specific role for intraflagellar transport genes during craniofacial development.

Elizabeth N Schock, Jaime N Struve, Ching-Fang Chang, Trevor J Williams, John Snedeker, Aria C Attia, Rolf W Stottmann, Samantha A Brugmann

Open access · goldAbstract read
In one paragraph

Article in PloS one, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 28 papers.

0numbers the graph read from it
0cells of the map it votes in
28citing 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

28 citing papers in PubMed, 38 citations in OpenAlex.

  1. Article
  2. Article
  3. Review
  4. Ciliary and Non-Ciliary Roles of IFT88 in Development and Diseases.International journal of molecular sciences · 2025
    Review
  5. Disruption of distal appendage protein CEP164 causes skeletal malformation in mice.Biochemical and biophysical research communications · 2024
    Article
  6. Actin cytoskeletal regulation of ciliogenesis in development and disease.Developmental dynamics : an official publication of the American Association of Anatomists · 2024
    Review
  7. Article
  8. Neural crest and periderm-specific requirements ofbioRxiv : the preprint server for biology · 2024
    Article
  9. Article
  10. Identification of a NovelMolecular syndromology · 2023
    Article
  11. A neural stem cell paradigm of pediatric hydrocephalus.Cerebral cortex (New York, N.Y. : 1991) · 2023
    Article
  12. Genetic Interaction ofJournal of developmental biology · 2022
    Article
  13. Article
  14. Article
  15. Article
  16. Article
  17. Article
  18. Investigating Primary Cilia during Peripheral Nervous System Formation.International journal of molecular sciences · 2021
    Article
  19. Article
  20. 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

8 authors at 2 institutions in 1 country.

Elizabeth N SchockDivision of Plastic Surgery, Department of Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, United States of America.ORCID http://orcid.org/0000-0002-3970-799X
Jaime N StruveDivision of Plastic Surgery, Department of Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, United States of America.
Ching-Fang ChangDivision of Plastic Surgery, Department of Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, United States of America.
Trevor J WilliamsDepartment of Craniofacial Biology, University of Colorado School of Dental Medicine, Aurora, Colorado, United States of America.
John SnedekerDivision of Human Genetics, Department of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, United States of America.
Aria C AttiaDivision of Human Genetics, Department of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, United States of America.ORCID http://orcid.org/0000-0002-1666-5395
Rolf W StottmannDivision of Developmental Biology, Department of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, United States of America.ORCID http://orcid.org/0000-0003-4512-6806
Samantha A BrugmannDivision of Plastic Surgery, Department of Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, United States of America.
Cincinnati Children's Hospital Medical Center · USUniversity of Colorado Denver · US

Funding

PREDOCTORAL TRAINING PROGRAM IN THE NEUROSCIENCEST32NS007453 · NINDS · UNIVERSITY OF CINCINNATI · PI HERMAN, JAMES P · 1997 to 2025
$4.0M
Role of Ectodermal Signals in Facial Prominence Outgrowth and DevelopmentR01DE019843 · NIDCR · UNIVERSITY OF COLORADO DENVER · PI WILLIAMS, TREVOR J · 2009 to 2013
$1.7M
The Role of Primary Cilia in Murine Craniofacial DevelopmentR01DE023804 · NIDCR · CINCINNATI CHILDRENS HOSP MED CTR · PI BRUGMANN, SAMANTHA A · 2014 to 2017
$1.6M
A genetic approach to defining the Ttc21b interactome in mammalian ciliopathiesR01GM112744 · NIGMS · CINCINNATI CHILDRENS HOSP MED CTR · PI STOTTMANN, ROLF W · 2015 to 2018
$1.2M
The role of ectodermal primary cilia in murine orofacial developmentF31DE025537 · NIDCR · CINCINNATI CHILDRENS HOSP MED CTR · PI SCHOCK, ELIZABETH (BETSY) · 2015 to 2017
$89k
NIDCR NIH HHS F31 DE025537NIDCR NIH HHS R01 DE019843NIDCR NIH HHS R01 DE023804NIGMS NIH HHS R01 GM112744NINDS NIH HHS T32 NS007453
6 · The paper itself

Abstract

Primary cilia are nearly ubiquitous, cellular projections that function to transduce molecular signals during development. Loss of functional primary cilia has a particularly profound effect on the developing craniofacial complex, causing several anomalies including craniosynostosis, micrognathia, midfacial dysplasia, cleft lip/palate and oral/dental defects. Development of the craniofacial complex is an intricate process that requires interactions between several different tissues including neural crest cells, neuroectoderm and surface ectoderm. To understand the tissue-specific requirements for primary cilia during craniofacial development we conditionally deleted three separate intraflagellar transport genes, Kif3a, Ift88 and Ttc21b with three distinct drivers, Wnt1-Cre, Crect and AP2-Cre which drive recombination in neural crest, surface ectoderm alone, and neural crest, surface ectoderm and neuroectoderm, respectively. We found that tissue-specific conditional loss of ciliary genes with different functions produces profoundly different facial phenotypes. Furthermore, analysis of basic cellular behaviors in these mutants suggests that loss of primary cilia in a distinct tissue has unique effects on development of adjacent tissues. Together, these data suggest specific spatiotemporal roles for intraflagellar transport genes and the primary cilium during craniofacial development.

Indexed as

Gene Expression Regulation, DevelopmentalAdaptor Proteins, Signal TransducingAnimalsCiliaCraniofacial AbnormalitiesFaceFemaleGene DeletionKinesinsMaleMiceNeural CrestNeural PlateSkullTumor Suppressor ProteinsAdaptor Proteins, Signal TransducingKif3a protein, mouseKinesinsTg737Rpw protein, mouseTtc21b protein, mouseTumor Suppressor Proteins

Identifiers

PMID28346501
PMCPMC5367710
OpenAlexW2600163047

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.