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

2018Journal article Open access
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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.accessioned2019-07-25T13:56:48Z
dc.date.available2019-07-25T13:56:48Z
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 diseases.pt_PT
dc.description.versioninfo:eu-repo/semantics/publishedVersionpt_PT
dc.identifier.doi10.1038/s41556-018-0132-1pt_PT
dc.identifier.urihttp://hdl.handle.net/10400.7/901
dc.language.isoengpt_PT
dc.subjectAnimalspt_PT
dc.subjectAnimals, Genetically Modifiedpt_PT
dc.subjectCaenorhabditis elegans Proteinspt_PT
dc.subjectCell Cycle Proteinspt_PT
dc.subjectCentriolespt_PT
dc.subjectCiliapt_PT
dc.subjectDrosophila Proteinspt_PT
dc.subjectDrosophila melanogasterpt_PT
dc.subjectFemalept_PT
dc.subjectFertilitypt_PT
dc.subjectMalept_PT
dc.subjectMicrotubule-Associated Proteinspt_PT
dc.subjectMutationpt_PT
dc.subjectNeuronspt_PT
dc.subjectPhenotypept_PT
dc.subjectSignal Transductionpt_PT
dc.subjectSmellpt_PT
dc.subjectSpermatozoapt_PT
dc.subjectTaxis Responsept_PT
dc.titleDifferential regulation of transition zone and centriole proteins contributes to ciliary base diversitypt_PT
dc.typejournal article
dspace.entity.typePublication
oaire.citation.endPage941pt_PT
oaire.citation.issue8pt_PT
oaire.citation.startPage928pt_PT
oaire.citation.volume20pt_PT
rcaap.rightsopenAccesspt_PT
rcaap.typearticlept_PT

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