Evidence map›Paper›PMID 32915243›Full record

ReviewCellular and molecular life sciences : CMLS2021

Motile cilia genetics and cell biology: big results from little mice.

Lance Lee, Lawrence E Ostrowski

Open access · greenAbstract readReview
In one paragraph

Review in Cellular and molecular life sciences : CMLS, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 36 papers.

0numbers the graph read from it
0cells of the map it votes in
36citing papers in PubMed
5.4field-weighted citation impact, top 4% 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

36 citing papers in PubMed, 53 citations in OpenAlex.

  1. Review
  2. Article
  3. Article
  4. Preclinical human models of primary ciliary dyskinesia.European respiratory review : an official journal of the European Respiratory Society · 2026
    Review
  5. Article
  6. Article
  7. Article
  8. Exome sequencing-based identification ofFrontiers in molecular biosciences · 2026
    Article
  9. Tppp3 determines basal body positioning and identity of respiratory cilia via microtubule assembly and sphingolipid homeostasis.Proceedings of the National Academy of Sciences of the United States of America · 2025
    Article
  10. Article
  11. EFCAB10 anchors AK8 to the radial spoke for proper ciliary motility.Proceedings of the National Academy of Sciences of the United States of America · 2025
    Article
  12. Article
  13. Article
  14. Review
  15. Ciliary and Non-Ciliary Roles of IFT88 in Development and Diseases.International journal of molecular sciences · 2025
    Review
  16. Article
  17. Development and Initial Characterization of Pigs withbioRxiv : the preprint server for biology · 2024
    Article
  18. Review
  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

2 authors at 2 institutions in 1 country.

Lance LeePediatrics and Rare Diseases Group, Sanford Research, Sioux Falls, SD, USA. lance.lee@sanfordhealth.org.ORCID http://orcid.org/0000-0002-8158-8699
Lawrence E OstrowskiMarsico Lung Institute/Cystic Fibrosis Center and Department of Pediatrics, University of North Carolina at Chapel Hill, Chapel Hill, NC, USA.ORCID http://orcid.org/0000-0002-7558-6603
Sanford Health · USUniversity of North Carolina at Chapel Hill · US

Funding

Functional Studies of Novel Genes Mutated in Primary Ciliary Dyskinesia II: Genotype to PhenotypeR01HL117836 · NHLBI · UNIV OF NORTH CAROLINA CHAPEL HILL · PI OSTROWSKI, LAWRENCE E · 2013 to 2023
$4.4M
NHLBI NIH HHS R01 HL117836NHLBI NIH HHS R01HL117836
6 · The paper itself

Abstract

Our understanding of motile cilia and their role in disease has increased tremendously over the last two decades, with critical information and insight coming from the analysis of mouse models. Motile cilia form on specific epithelial cell types and typically beat in a coordinated, whip-like manner to facilitate the flow and clearance of fluids along the cell surface. Defects in formation and function of motile cilia result in primary ciliary dyskinesia (PCD), a genetically heterogeneous disorder with a well-characterized phenotype but no effective treatment. A number of model systems, ranging from unicellular eukaryotes to mammals, have provided information about the genetics, biochemistry, and structure of motile cilia. However, with remarkable resources available for genetic manipulation and developmental, pathological, and physiological analysis of phenotype, the mouse has risen to the forefront of understanding mammalian motile cilia and modeling PCD. This is evidenced by a large number of relevant mouse lines and an extensive body of genetic and phenotypic data. More recently, application of innovative cell biological techniques to these models has enabled substantial advancement in elucidating the molecular and cellular mechanisms underlying the biogenesis and function of mammalian motile cilia. In this article, we will review genetic and cell biological studies of motile cilia in mouse models and their contributions to our understanding of motile cilia and PCD pathogenesis.

Indexed as

AnimalsArmadillo Domain ProteinsAxonemeCiliaCiliary Motility DisordersDisease Models, AnimalDyneinsMiceMicrotubule-Associated ProteinsProtein BindingArmadillo Domain ProteinsARMC4 protein, mouseDNAAF1 protein, mouseDyneinsMicrotubule-Associated ProteinsCiliaMotile ciliaMouseMucociliary clearancePCDPrimary ciliary dyskinesia

Identifiers

PMID32915243
PMCPMC7902362
OpenAlexW3085007210

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.