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A phylogenetic profiling approach identifies novel ciliogenesis genes in Drosophila and C. elegans

Cilia are cellular projections that perform sensory and motile functions in eukaryotic cells. A defining feature of cilia is that they are evolutionarily ancient, yet not universally conserved. In this study, we have used the resulting presence and absence pattern in the genomes of diverse eukaryote...

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Autores principales: Dobbelaere, Jeroen, Su, Tiffany Y, Erdi, Balazs, Schleiffer, Alexander, Dammermann, Alexander
Formato: Online Artículo Texto
Lenguaje:English
Publicado: John Wiley and Sons Inc. 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10425847/
https://www.ncbi.nlm.nih.gov/pubmed/37317646
http://dx.doi.org/10.15252/embj.2023113616
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author Dobbelaere, Jeroen
Su, Tiffany Y
Erdi, Balazs
Schleiffer, Alexander
Dammermann, Alexander
author_facet Dobbelaere, Jeroen
Su, Tiffany Y
Erdi, Balazs
Schleiffer, Alexander
Dammermann, Alexander
author_sort Dobbelaere, Jeroen
collection PubMed
description Cilia are cellular projections that perform sensory and motile functions in eukaryotic cells. A defining feature of cilia is that they are evolutionarily ancient, yet not universally conserved. In this study, we have used the resulting presence and absence pattern in the genomes of diverse eukaryotes to identify a set of 386 human genes associated with cilium assembly or motility. Comprehensive tissue‐specific RNAi in Drosophila and mutant analysis in C. elegans revealed signature ciliary defects for 70–80% of novel genes, a percentage similar to that for known genes within the cluster. Further characterization identified different phenotypic classes, including a set of genes related to the cartwheel component Bld10/CEP135 and two highly conserved regulators of cilium biogenesis. We propose this dataset defines the core set of genes required for cilium assembly and motility across eukaryotes and presents a valuable resource for future studies of cilium biology and associated disorders.
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spelling pubmed-104258472023-08-16 A phylogenetic profiling approach identifies novel ciliogenesis genes in Drosophila and C. elegans Dobbelaere, Jeroen Su, Tiffany Y Erdi, Balazs Schleiffer, Alexander Dammermann, Alexander EMBO J Resource Cilia are cellular projections that perform sensory and motile functions in eukaryotic cells. A defining feature of cilia is that they are evolutionarily ancient, yet not universally conserved. In this study, we have used the resulting presence and absence pattern in the genomes of diverse eukaryotes to identify a set of 386 human genes associated with cilium assembly or motility. Comprehensive tissue‐specific RNAi in Drosophila and mutant analysis in C. elegans revealed signature ciliary defects for 70–80% of novel genes, a percentage similar to that for known genes within the cluster. Further characterization identified different phenotypic classes, including a set of genes related to the cartwheel component Bld10/CEP135 and two highly conserved regulators of cilium biogenesis. We propose this dataset defines the core set of genes required for cilium assembly and motility across eukaryotes and presents a valuable resource for future studies of cilium biology and associated disorders. John Wiley and Sons Inc. 2023-06-15 /pmc/articles/PMC10425847/ /pubmed/37317646 http://dx.doi.org/10.15252/embj.2023113616 Text en © 2023 The Authors. Published under the terms of the CC BY 4.0 license https://creativecommons.org/licenses/by/4.0/This is an open access article under the terms of the http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited.
spellingShingle Resource
Dobbelaere, Jeroen
Su, Tiffany Y
Erdi, Balazs
Schleiffer, Alexander
Dammermann, Alexander
A phylogenetic profiling approach identifies novel ciliogenesis genes in Drosophila and C. elegans
title A phylogenetic profiling approach identifies novel ciliogenesis genes in Drosophila and C. elegans
title_full A phylogenetic profiling approach identifies novel ciliogenesis genes in Drosophila and C. elegans
title_fullStr A phylogenetic profiling approach identifies novel ciliogenesis genes in Drosophila and C. elegans
title_full_unstemmed A phylogenetic profiling approach identifies novel ciliogenesis genes in Drosophila and C. elegans
title_short A phylogenetic profiling approach identifies novel ciliogenesis genes in Drosophila and C. elegans
title_sort phylogenetic profiling approach identifies novel ciliogenesis genes in drosophila and c. elegans
topic Resource
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10425847/
https://www.ncbi.nlm.nih.gov/pubmed/37317646
http://dx.doi.org/10.15252/embj.2023113616
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