Regulation of ciliary membrane protein trafficking and signalling by kinesin motor proteins

Morthorst, Stine Kjær; Christensen, Søren Tvorup; Pedersen, Lotte Bang

doi:10.1111/febs.14583

Cited by 49Genetic and Kidney Cyst DiseasesCystic Fibrosis Research AdvancesMicrotubule and mitosis dynamics

Regulation of ciliary membrane protein trafficking and signalling by kinesin motor proteins

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Morthorst, Stine Kjær, Christensen, Søren Tvorup, Pedersen, Lotte Bang·2018·The FEBS Journal

DOI 10.1111/febs.14583

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Abstract

Primary cilia are antenna‐like sensory organelles that regulate a substantial number of cellular signalling pathways in vertebrates, both during embryonic development as well as in adulthood, and mutations in genes coding for ciliary proteins are causative of an expanding group of pleiotropic diseases known as ciliopathies. Cilia consist of a microtubule‐based axoneme core, which is subtended by a basal body and covered by a bilayer lipid membrane of unique protein and lipid composition. Cilia are dynamic organelles, and the ability of cells to regulate ciliary protein and lipid content in response to specific cellular and environmental cues is crucial for balancing ciliary signalling output. Here we discuss mechanisms involved in regulation of ciliary membrane protein trafficking and signalling, with main focus on kinesin‐2 and kinesin‐3 family members.

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Many studies in different organisms confirmed a universal requirement for heterotrimeric kinesin-2 and cytoplasmic dynein 2 in IFT, but some organisms also use accessory kinesins to regulate aspects of ciliary assembly or function.
“A large number of studies in different organisms subsequently confirmed a universal requirement for these motors in IFT, but also demonstrated that some organisms additionally use ‘accessory’ kinesins to regulate certain aspects of ciliary assembly or function 23, 39.”
From Cellular signalling by primary cilia in development, organ function and disease (2019) · cites this paper as [23]
The “Intraflagellar transport” section (page not available) describes kinesin-2 and dynein 2 as IFT motors, but says mutations in their subunits usually cause severe ciliary defects in most organisms studied—not that their requirement is universal. The “Role of kinesin-2 motors in axoneme extension” section (page not available) describes additional kinesin-2 motors with overlapping or partially redundant roles in some organisms, supporting the accessory-motor part of the claim. (LLM, conf=0.87)
Experimental — accuracy not yet measured
The mechanisms that control the balanced output of ciliary signaling described in this Review might also apply to other pathways such as Hippo and Notch signaling pathways or those regulated by receptors of extracellular matrix molecules, which also have been linked to primary cilia in various cell types and tissues.
“The mechanisms that control the balanced output of ciliary signalling described in this Review might also apply to other pathways such as Hippo and Notch signalling pathways or those regulated by receptors of extracellular matrix molecules, which also have been linked to primary cilia in various cell types and tissues 23, 291, 292.”
From Cellular signalling by primary cilia in development, organ function and disease (2019) · cites this paper as [23]
The Introduction links Notch and Hippo signalling to primary cilia, and the TGF-β/BMP signalling section describes ciliary control of balanced signalling output. The paper does not substantiate the additional claim that pathways regulated by receptors of extracellular-matrix molecules are linked to primary cilia in various cell types and tissues. (LLM, conf=0.84)
Experimental — accuracy not yet measured
Vesicular transport pathways deliver specific receptors or signalling molecules from the Golgi or recycling endosome to the ciliary base, where vesicles are exocytosed.
“These include vesicular transport pathways that target specific receptors or signalling molecules from the Golgi or recycling endosome to the ciliary base where vesicles are exocytosed 22, 23,”
From Cellular signalling by primary cilia in development, organ function and disease (2019) · cites this paper as [23]

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Cellular signalling by primary cilia in development, organ function and disease
Anvarian, Z., Mykytyn, K., Mukhopadhyay, S., Pedersen, L. B., & Christensen, S. T. (2019) · Nature Reviews Nephrology · doi:10.1038/s41581-019-0116-9 · Cited by 845 · see citing works
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Primary cilia as dynamic and diverse signalling hubs in development and disease
Mill, P., Christensen, S. T., & Pedersen, L. B. (2023) · Nature Reviews Genetics · doi:10.1038/s41576-023-00587-9 · Cited by 417 · see citing works
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Authors: Morthorst, S. K., Christensen, S. T., & Pedersen, L. B.
Affiliations: University of Copenhagen (DK)
Corresponding author: Pedersen, Lotte Bang
ORCID: Morthorst, Stine Kjær
Christensen, Søren Tvorup
Pedersen, Lotte Bang
Venue: The FEBS Journal
Publisher: Wiley
Published: 2018-06-27
Volume: 285
Issue: 24
Pages: 4535-4564
ISSN: 1742-464X (ISSN-L)
1742-4658
DOI: 10.1111/febs.14583
OpenAlex: W2807817315
Cited by: 49
References: 324
Citations by year: 2026: 4 · 2025: 3 · 2024: 5 · 2023: 4 · 2022: 8 · 2021: 9 · 2020: 5 · 2019: 9 · 2018: 2
FWCI: 0.88
Type: journal article
Language: en
Open access: bronze
doi.org
Licence: onlinelibrary.wiley.com/termsAndConditions#vor (vor)
Locations: FEBS Journal (publishedVersion)
PubMed (publishedVersion)
Research at the University of Copenhagen (University of Copenhagen) (submittedVersion)
Funders: Novo Nordisk Fonden (NNF15OC0016886)
Kræftens Bekæmpelse (R146‐A9590‐16‐S2)
Carlsbergfondet (CF15‐0425)
Hartmann Fonden (A311662)
Københavns Universitet
Topics: Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetic and Kidney Cyst Diseases
Health Sciences › Medicine › Pulmonary and Respiratory Medicine › Cystic Fibrosis Research Advances
Life Sciences › Biochemistry, Genetics and Molecular Biology › Cell Biology › Microtubule and mitosis dynamics
Keywords: primary cilia, kinesin-2, kinesin-3, ciliopathies, basal bodies, axoneme, microtubule transport
MeSH terms: Animals, Biological Transport, Cell Movement, Cilia, Flagella, Humans, Membrane Proteins, Signal Transduction, Kinesins

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Regulation of ciliary membrane protein trafficking and signalling by kinesin motor proteins

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