Differential regulation of transition zone and centriole proteins contributes to ciliary base diversity

Detalhes bibliográficos
Autor(a) principal: Jana, Swadhin Chandra
Data de Publicação: 2018
Outros Autores: Mendonça, Susana, Machado, Pedro, Werner, Sascha, Rocha, Jaqueline, Pereira, António, Maiato, Helder, Bettencourt-Dias, Mónica
Tipo de documento: Artigo
Idioma: eng
Título da fonte: Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos)
Texto Completo: http://hdl.handle.net/10400.14/28448
Resumo: Cilia are evolutionarily conserved structures with many sensory and motility-related functions. The ciliary base, composed of the basal body and the transition zone, is critical for cilia assembly and function, but its contribution to cilia diversity remains unknown. Hence, we generated a high-resolution structural and biochemical atlas of the ciliary base of four functionally distinct neuronal and sperm cilia types within an organism, Drosophila melanogaster. We uncovered a common scaffold and diverse structures associated with different localization of 15 evolutionarily conserved components. Furthermore, CEP290 (also known as NPHP6) is involved in the formation of highly diverse transition zone links. In addition, the cartwheel components SAS6 and ANA2 (also known as STIL) have an underappreciated role in basal body elongation, which depends on BLD10 (also known as CEP135). The differential expression of these cartwheel components contributes to diversity in basal body length. Our results offer a plausible explanation to how mutations in conserved ciliary base components lead to tissue-specific diseases
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spelling Differential regulation of transition zone and centriole proteins contributes to ciliary base diversityCilia are evolutionarily conserved structures with many sensory and motility-related functions. The ciliary base, composed of the basal body and the transition zone, is critical for cilia assembly and function, but its contribution to cilia diversity remains unknown. Hence, we generated a high-resolution structural and biochemical atlas of the ciliary base of four functionally distinct neuronal and sperm cilia types within an organism, Drosophila melanogaster. We uncovered a common scaffold and diverse structures associated with different localization of 15 evolutionarily conserved components. Furthermore, CEP290 (also known as NPHP6) is involved in the formation of highly diverse transition zone links. In addition, the cartwheel components SAS6 and ANA2 (also known as STIL) have an underappreciated role in basal body elongation, which depends on BLD10 (also known as CEP135). The differential expression of these cartwheel components contributes to diversity in basal body length. Our results offer a plausible explanation to how mutations in conserved ciliary base components lead to tissue-specific diseasesSpringerVeritati - Repositório Institucional da Universidade Católica PortuguesaJana, Swadhin ChandraMendonça, SusanaMachado, PedroWerner, SaschaRocha, JaquelinePereira, AntónioMaiato, HelderBettencourt-Dias, Mónica2019-10-21T13:52:36Z20182018-01-01T00:00:00Zinfo:eu-repo/semantics/publishedVersioninfo:eu-repo/semantics/articleapplication/pdfhttp://hdl.handle.net/10400.14/28448engJana, S. C., Mendonça, S., Machado, P., Werner, S., Rocha, J., Pereira, A., Maiato, H., Bettencourt-Dias, M. (2018). Differential regulation of transition zone and centriole proteins contributes to ciliary base diversity. Nature Cell Biology, 20(8), 928–9411476-467910.1038/s41556-018-0132-11476-46798504993273530013109000440057000012info:eu-repo/semantics/openAccessreponame:Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos)instname:Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informaçãoinstacron:RCAAP2023-09-19T01:41:20Zoai:repositorio.ucp.pt:10400.14/28448Portal AgregadorONGhttps://www.rcaap.pt/oai/openaireopendoar:71602024-03-19T18:22:49.080779Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos) - Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informaçãofalse
dc.title.none.fl_str_mv Differential regulation of transition zone and centriole proteins contributes to ciliary base diversity
title Differential regulation of transition zone and centriole proteins contributes to ciliary base diversity
spellingShingle Differential regulation of transition zone and centriole proteins contributes to ciliary base diversity
Jana, Swadhin Chandra
title_short Differential regulation of transition zone and centriole proteins contributes to ciliary base diversity
title_full Differential regulation of transition zone and centriole proteins contributes to ciliary base diversity
title_fullStr Differential regulation of transition zone and centriole proteins contributes to ciliary base diversity
title_full_unstemmed Differential regulation of transition zone and centriole proteins contributes to ciliary base diversity
title_sort Differential regulation of transition zone and centriole proteins contributes to ciliary base diversity
author Jana, Swadhin Chandra
author_facet Jana, Swadhin Chandra
Mendonça, Susana
Machado, Pedro
Werner, Sascha
Rocha, Jaqueline
Pereira, António
Maiato, Helder
Bettencourt-Dias, Mónica
author_role author
author2 Mendonça, Susana
Machado, Pedro
Werner, Sascha
Rocha, Jaqueline
Pereira, António
Maiato, Helder
Bettencourt-Dias, Mónica
author2_role author
author
author
author
author
author
author
dc.contributor.none.fl_str_mv Veritati - Repositório Institucional da Universidade Católica Portuguesa
dc.contributor.author.fl_str_mv Jana, Swadhin Chandra
Mendonça, Susana
Machado, Pedro
Werner, Sascha
Rocha, Jaqueline
Pereira, António
Maiato, Helder
Bettencourt-Dias, Mónica
description Cilia are evolutionarily conserved structures with many sensory and motility-related functions. The ciliary base, composed of the basal body and the transition zone, is critical for cilia assembly and function, but its contribution to cilia diversity remains unknown. Hence, we generated a high-resolution structural and biochemical atlas of the ciliary base of four functionally distinct neuronal and sperm cilia types within an organism, Drosophila melanogaster. We uncovered a common scaffold and diverse structures associated with different localization of 15 evolutionarily conserved components. Furthermore, CEP290 (also known as NPHP6) is involved in the formation of highly diverse transition zone links. In addition, the cartwheel components SAS6 and ANA2 (also known as STIL) have an underappreciated role in basal body elongation, which depends on BLD10 (also known as CEP135). The differential expression of these cartwheel components contributes to diversity in basal body length. Our results offer a plausible explanation to how mutations in conserved ciliary base components lead to tissue-specific diseases
publishDate 2018
dc.date.none.fl_str_mv 2018
2018-01-01T00:00:00Z
2019-10-21T13:52:36Z
dc.type.status.fl_str_mv info:eu-repo/semantics/publishedVersion
dc.type.driver.fl_str_mv info:eu-repo/semantics/article
format article
status_str publishedVersion
dc.identifier.uri.fl_str_mv http://hdl.handle.net/10400.14/28448
url http://hdl.handle.net/10400.14/28448
dc.language.iso.fl_str_mv eng
language eng
dc.relation.none.fl_str_mv Jana, S. C., Mendonça, S., Machado, P., Werner, S., Rocha, J., Pereira, A., Maiato, H., Bettencourt-Dias, M. (2018). Differential regulation of transition zone and centriole proteins contributes to ciliary base diversity. Nature Cell Biology, 20(8), 928–941
1476-4679
10.1038/s41556-018-0132-1
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dc.publisher.none.fl_str_mv Springer
publisher.none.fl_str_mv Springer
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