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

Swadhin Chandra Jana*, Susana Mendonça, Pedro Machado, Sascha Werner, Jaqueline Rocha, António Pereira, Helder Maiato, Mónica Bettencourt-Dias

*Corresponding author for this work

Research output: Contribution to journalArticlepeer-review

61 Citations (Scopus)
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Abstract

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.
Original languageEnglish
Pages (from-to)928-941
Number of pages14
JournalNature Cell Biology
Volume20
Issue number8
DOIs
Publication statusPublished - 1 Aug 2018

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