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

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

http://hdl.handle.net/10400.7/901

Use this identifier to reference this record.

Name:	Description:	Size:	Format:
Janaetal.pdf		8.91 MB	Adobe PDF	Download

Send Feedback

Authors

Jana, Swadhin Chandra

Bettencourt-Dias, Mónica

Abstract(s)

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.

Keywords

Animals Animals, Genetically Modified Caenorhabditis elegans Proteins Cell Cycle Proteins Centrioles Cilia Drosophila Proteins Drosophila melanogaster Female Fertility Male Microtubule-Associated Proteins Mutation Neurons Phenotype Signal Transduction Smell Spermatozoa Taxis Response