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

dc.contributor.authorJana, Swadhin Chandra
dc.contributor.authorMendonça, Susana
dc.contributor.authorMachado, Pedro
dc.contributor.authorWerner, Sascha
dc.contributor.authorRocha, Jaqueline
dc.contributor.authorPereira, António
dc.contributor.authorMaiato, Helder
dc.contributor.authorBettencourt-Dias, Mónica
dc.date.accessioned2019-10-21T13:52:36Z
dc.date.available2019-10-21T13:52:36Z
dc.date.issued2018
dc.description.abstractCilia 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 diseasespt_PT
dc.description.versioninfo:eu-repo/semantics/acceptedVersionpt_PT
dc.identifier.citationJana, 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), 928–941pt_PT
dc.identifier.doi10.1038/s41556-018-0132-1pt_PT
dc.identifier.eid85049932735
dc.identifier.eissn1476-4679
dc.identifier.issn1476-4679
dc.identifier.pmid30013109
dc.identifier.urihttp://hdl.handle.net/10400.14/28448
dc.identifier.wos000440057000012
dc.language.isoengpt_PT
dc.peerreviewedyespt_PT
dc.publisherSpringerpt_PT
dc.relationERC-681443-CODECHECK
dc.relationERC-261344-CentrioleStructNumb
dc.relationDYNAMICS AND MAINTENANCE OF CILIA COMPONENTS
dc.rights.urihttps://creativecommons.org/licenses/by/4.0
dc.titleDifferential regulation of transition zone and centriole proteins contributes to ciliary base diversitypt_PT
dc.typejournal article
dspace.entity.typePublication
oaire.awardTitleDYNAMICS AND MAINTENANCE OF CILIA COMPONENTS
oaire.awardURIinfo:eu-repo/grantAgreement/FCT/SFRH/SFRH%2FBPD%2F87479%2F2012/PT
oaire.awardURIinfo:eu-repo/grantAgreement/FCT//SFRH%2FBD%2F52176%2F2013/PT
oaire.citation.endPage941
oaire.citation.issue8
oaire.citation.startPage928
oaire.citation.titleNature Cell Biologypt_PT
oaire.citation.volume20
oaire.fundingStreamSFRH
person.familyNameJana
person.familyNameMendonça
person.familyNameWerner
person.familyNameRocha
person.familyNamePereira
person.familyNameBettencourt-Dias
person.givenNameSwadhin
person.givenNameSusana
person.givenNameSascha
person.givenNameJaqueline
person.givenNameAntónio
person.givenNameMonica
person.identifier.ciencia-id8116-CD55-5B6C
person.identifier.ciencia-idA215-4435-A06B
person.identifier.ciencia-id721C-C3E4-6A38
person.identifier.ciencia-id4016-40EA-B364
person.identifier.ciencia-id9C1E-886C-1169
person.identifier.orcid0000-0002-8311-3849
person.identifier.orcid0000-0001-9566-4026
person.identifier.orcid0000-0001-8350-2417
person.identifier.orcid0000-0003-1651-9483
person.identifier.orcid0000-0002-1053-8715
person.identifier.orcid0000-0003-1987-5598
person.identifier.ridK-3850-2014
person.identifier.ridB-5176-2015
person.identifier.scopus-author-id35269080600
person.identifier.scopus-author-id57189463026
person.identifier.scopus-author-id57113035100
person.identifier.scopus-author-id55506297800
person.identifier.scopus-author-id16318673000
project.funder.identifierhttp://doi.org/10.13039/501100001871
project.funder.identifierhttp://doi.org/10.13039/501100001871
project.funder.nameFundação para a Ciência e a Tecnologia
project.funder.nameFundação para a Ciência e a Tecnologia
rcaap.rightsopenAccesspt_PT
rcaap.typearticlept_PT
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