Cited by 44Genetic and Kidney Cyst DiseasesCystic Fibrosis Research AdvancesRetinal Development and Disorders

Compartmentalization of Photoreceptor Sensory Cilia

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Barnes, Cassandra L., Malhotra, Himanshu, Calvert, Peter D.·2021·Frontiers in Cell and Developmental Biology

Abstract

Functional compartmentalization of cells is a universal strategy for segregating processes that require specific components, undergo regulation by modulating concentrations of those components, or that would be detrimental to other processes. Primary cilia are hair-like organelles that project from the apical plasma membranes of epithelial cells where they serve as exclusive compartments for sensing physical and chemical signals in the environment. As such, molecules involved in signal transduction are enriched within cilia and regulating their ciliary concentrations allows adaptation to the environmental stimuli. The highly efficient organization of primary cilia has been co-opted by major sensory neurons, olfactory cells and the photoreceptor neurons that underlie vision. The mechanisms underlying compartmentalization of cilia are an area of intense current research. Recent findings have revealed similarities and differences in molecular mechanisms of ciliary protein enrichment and its regulation among primary cilia and sensory cilia. Here we discuss the physiological demands on photoreceptors that have driven their evolution into neurons that rely on a highly specialized cilium for signaling changes in light intensity. We explore what is known and what is not known about how that specialization appears to have driven unique mechanisms for photoreceptor protein and membrane compartmentalization.

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Crossref · 10.3389/fcell.2021.636737

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  • Retinal dystrophy represents a common clinical phenotype in several non-motile ciliopathies.

    “Retinal dystrophy represents a common clinical phenotype in several non-motile ciliopathies12,73,74.”

    From The inner junction protein CFAP20 functions in motile and non-motile cilia and is critical for vision (2022) · cites this paper as [74]

    The section “The BBSome in Photoreceptors” states that Bardet–Biedl syndrome is a ciliopathy that leads to retinal degeneration and that almost all patients develop retinitis pigmentosa. This supports retinal dystrophy as a common phenotype in that syndrome, but the passages do not establish its prevalence across several non-motile ciliopathies. (LLM, conf=0.82)

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Research works printed but not linked: 1

  1. Potter V. L. Robichaux M. A. Moye A. R. Wensel T. G. (2020). Superresolution microscopy reveals photoreceptor-specific subciliary location and function of Cep290. bioRxiv [Preprint]. 10.1101/2020.10.28.357806 PMC8564900 34520396 · DOI

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Authors
Barnes, C. L., Malhotra, H., & Calvert, P. D.
Affiliations
SUNY Upstate Medical University (US)
Corresponding author
Calvert, Peter D.
Venue
Frontiers in Cell and Developmental Biology
Published
2021-02-04
Volume
9
Pages
636737-636737
ISSN
2296-634X (ISSN-L)
OpenAlex
W3127000513
Cited by
44
References
317
Citations by year
2026: 7 · 2025: 8 · 2024: 10 · 2023: 8 · 2022: 6 · 2021: 5
FWCI
1.07
Type
journal article
Language
en
Open access
Locations
  • Frontiers in Cell and Developmental Biology (publishedVersion, cc-by)
  • PubMed Central (publishedVersion, cc-by)
  • PubMed (publishedVersion)
  • DOAJ (DOAJ: Directory of Open Access Journals) (submittedVersion, cc-by-sa)
Funders
  • National Eye Institute (R01 EY028303, R01EY018421)
  • Research to Prevent Blindness (Stein Innovation Award)
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 › Molecular Biology › Retinal Development and Disorders
Keywords
photoreceptors, primary cilia, sensory cilia, signal transduction, light signaling, olfactory sensory neurons

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