Evidence map›Paper›PMID 30446775›Full record

ArticleCellular and molecular life sciences : CMLS2019

Bardet-Biedl Syndrome proteins regulate cilia disassembly during tissue maturation.

Sarita Rani Patnaik, Viola Kretschmer, Lena Brücker, Sandra Schneider, Ann-Kathrin Volz, Liliana Del Rocio Oancea-Castillo, Helen Louise May-Simera

Open access · greenAbstract read
In one paragraph

Article in Cellular and molecular life sciences : CMLS, 2019. 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
3.3field-weighted citation impact, top 7% 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. Article
  2. Taking Down the Primary Cilium: Pathways for Disassembly in Differentiating Cells.BioEssays : news and reviews in molecular, cellular and developmental biology · 2025
    Review
  3. Complement C3 Activation in the Human Retinal Pigment Epithelium.Investigative ophthalmology & visual science · 2025
    Article
  4. Article
  5. Review
  6. Histone Deacetylase 6 (HDAC6) in Ciliopathies: Emerging Insights and Therapeutic Implications.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2025
    Review
  7. Article
  8. Role of Primary Cilia in the Eye.Advances in experimental medicine and biology · 2025
    Review
  9. Article
  10. Article
  11. Article
  12. Review
  13. Article
  14. Article
  15. Article
  16. Review
  17. Article
  18. Article
  19. WGS Revealed NovelInternational journal of molecular sciences · 2023
    Article
  20. The complex relationship of Wnt-signaling pathways and cilia.Current topics in developmental biology · 2023
    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

7 authors at 1 institution in 1 country.

Sarita Rani PatnaikCilia Cell Biology, Institute of Molecular Physiology, Johannes-Gutenberg University, 55128, Mainz, Germany.
Viola KretschmerCilia Cell Biology, Institute of Molecular Physiology, Johannes-Gutenberg University, 55128, Mainz, Germany.
Lena BrückerCilia Cell Biology, Institute of Molecular Physiology, Johannes-Gutenberg University, 55128, Mainz, Germany.
Sandra SchneiderCilia Cell Biology, Institute of Molecular Physiology, Johannes-Gutenberg University, 55128, Mainz, Germany.ORCID http://orcid.org/0000-0001-8494-1788
Ann-Kathrin VolzCilia Cell Biology, Institute of Molecular Physiology, Johannes-Gutenberg University, 55128, Mainz, Germany.
Liliana Del Rocio Oancea-CastilloInstitute of Developmental Biology and Neurobiology, Johannes-Gutenberg University, 55128, Mainz, Germany.
Helen Louise May-SimeraCilia Cell Biology, Institute of Molecular Physiology, Johannes-Gutenberg University, 55128, Mainz, Germany. hmaysime@uni-mainz.de.ORCID http://orcid.org/0000-0002-9477-4036
Johannes Gutenberg University Mainz · DE

Funding

Alexander von Humboldt-Stiftung Sofija Kovalevskaja Award
6 · The paper itself

Abstract

Primary cilia are conserved organelles that mediate cellular communication crucial for organogenesis and homeostasis in numerous tissues. The retinal pigment epithelium (RPE) is a ciliated monolayer in the eye that borders the retina and is vital for visual function. Maturation of the RPE is absolutely critical for visual function and the role of the primary cilium in this process has been largely ignored to date. We show that primary cilia are transiently present during RPE development and that as the RPE matures, primary cilia retract, and gene expression of ciliary disassembly components decline. We observe that ciliary-associated BBS proteins protect against HDAC6-mediated ciliary disassembly via their recruitment of Inversin to the base of the primary cilium. Inhibition of ciliary disassembly components was able to rescue ciliary length defects in BBS deficient cells. This consequently affects ciliary regulation of Wnt signaling. Our results shed light onto the mechanisms by which cilia-mediated signaling facilitates tissue maturation.

Indexed as

AnimalsCiliaCytoskeletal ProteinsEmbryo, MammalianGene Expression Regulation, DevelopmentalGroup II ChaperoninsHEK293 CellsHistone Deacetylase 6HumansMice, Inbred C57BLMice, KnockoutMicrotubule-Associated ProteinsRetinal Pigment EpitheliumRNA InterferenceWnt Signaling PathwayCytoskeletal ProteinsGroup II ChaperoninsHdac6 protein, mouseHistone Deacetylase 6Microtubule-Associated ProteinsMkks protein, mouseTtc8 protein, mouseCiliopathyProteasomal degradationRetinal dystrophySignaling inhibitorsSignaling pathways

Identifiers

PMID30446775
PMCPMC11105770
OpenAlexW2900917763

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.