ArticleMolecular biology of the cell2011
The Pcdp1 complex coordinates the activity of dynein isoforms to produce wild-type ciliary motility.
Article in Molecular biology of the cell, 2011. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 17 papers.
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Who cites it
17 citing papers in PubMed, 28 citations in OpenAlex.
- Airway ciliary microenvironment responses in mice with primary ciliary dyskinesia and central pair apparatus defects.Scientific reports · 2024Article
- Lack of CFAP54 causes primary ciliary dyskinesia in a mouse model and human patients.Frontiers of medicine · 2023Article
- Concurrent loss of ciliary genesRoyal Society open science · 2023Article
- Cryo-EM structure of an active central apparatus.Nature structural & molecular biology · 2022Article
- Structural organization of the C1b projection within the ciliary central apparatus.Journal of cell science · 2021Article
- Analysis of the sperm flagellar axoneme using gene-modified mice.Experimental animals · 2020Review
- Identification of Potential Therapeutic Gene Markers in Nasopharyngeal Carcinoma Based on Bioinformatics Analysis.Clinical and translational science · 2020Article
- Ammonia Exposure Induced Cilia Dysfunction of Nasal Mucosa in the Piglets.BioMed research international · 2020Article
- Structural organization of the C1a-e-c supercomplex within the ciliary central apparatus.The Journal of cell biology · 2019Article
- Review
- Proteome of the central apparatus of a ciliary axoneme.The Journal of cell biology · 2019Article
- The Central Apparatus of Cilia and Eukaryotic Flagella.Cold Spring Harbor perspectives in biology · 2017Review
- Mammalian axoneme central pair complex proteins: Broader roles revealed by gene knockout phenotypes.Cytoskeleton (Hoboken, N.J.) · 2016Review
- CFAP54 is required for proper ciliary motility and assembly of the central pair apparatus in mice.Molecular biology of the cell · 2015Article
- A FAP46 mutant provides new insights into the function and assembly of the C1d complex of the ciliary central apparatus.Journal of cell science · 2012Article
- The CSC connects three major axonemal complexes involved in dynein regulation.Molecular biology of the cell · 2012Article
- Analyses of functional domains within the PF6 protein of the central apparatus reveal a role for PF6 sub-complex members in regulating flagellar beat frequency.Cytoskeleton (Hoboken, N.J.) · 2012Article
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2 authors at 2 institutions in 1 country.
Funding
Abstract
Generating the complex waveforms characteristic of beating cilia requires the coordinated activity of multiple dynein isoforms anchored to the axoneme. We previously identified a complex associated with the C1d projection of the central apparatus that includes primary ciliary dyskinesia protein 1 (Pcdp1). Reduced expression of complex members results in severe motility defects, indicating that C1d is essential for wild-type ciliary beating. To define a mechanism for Pcdp1/C1d regulation of motility, we took a functional and structural approach combined with mutants lacking C1d and distinct subsets of dynein arms. Unlike mutants completely lacking the central apparatus, dynein-driven microtubule sliding velocities are wild type in C1d- defective mutants. However, coordination of dynein activity among microtubule doublets is severely disrupted. Remarkably, mutations in either outer or inner dynein arm restore motility to mutants lacking C1d, although waveforms and beat frequency differ depending on which isoform is mutated. These results define a unique role for C1d in coordinating the activity of specific dynein isoforms to control ciliary motility.
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