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Investigation of a novel cilia-related gene K04F10.2/KIAA0556 in C. elegans

Multiple proteomics and genomics approaches have been used to identify the molecular parts list of cilia and flagella. However, the specific cilia-related functions of many of these components remain unknown. Previously, K04F10.2 was identified as a candidate cilia-related gene in C. elegans, exhibiting specific expression in ciliated sensory neurons. We now show that GFP-tagged K04F10.2 is highly enriched at the transition zone (TZ) compartment at the base of the ciliary axoneme, and possibly to the more proximal transition fiber/basal body region. Fluorescence microscopy and transmission electron microscopy show that K04F10.2 null mutants possess grossly normal cilium structure and ultrastructure. In contrast, wild-type worms overexpressing a K04F10.2::gfp transgene possess cilium integrity defects such as a dye-filling abnormality (Dyf) and phasmid cilia that are frequently short and abnormally separated. In addition, phasmid neuronal dendrites are abnormally short in these worms. Since GFP-tagged K04F10.2 is enriched at the TZ, we examined possible genetic relationships with known TZ-associated ciliary disease genes such as those causing Meckel-Gruber syndrome (MKS) and Nephronophthisis (NPHP). Unlike the synthetic Dyf (SynDyf) phenotypes known for alleles of various MKS and NPHP genes, no such phenotype was observed in K04F10.2;mks-5 and K04F10.2;nphp-4 double mutants. However, K04F10.2 was found to synthetically interact (SynDyf) with Joubert syndrome-associated Arl13b/arl-13, which interestingly does not localise at the TZ. Together, these data implicate K04F10.2 as a novel TZ-enriched protein with functions that are distinct from canonical MKS and NPHP module components.

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Correspondence to A Sanders.

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This article is published under license to BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

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Sanders, A., Cevik, S., Kida, K. et al. Investigation of a novel cilia-related gene K04F10.2/KIAA0556 in C. elegans. Cilia 1 (Suppl 1), P43 (2012).

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