Regulation of flagellar motility by the conserved flagellar protein CG34110/Ccdc135/FAP50

• Published in: Molecular biology of the cell Vol. 22; no. 7; pp. 976 - 987
• Main Authors: Yang, Yong, Cochran, Deborah A, Gargano, Mary D, King, Iryna, Samhat, Nayef K, Burger, Benjain P, Sabourin, Katherine R, Hou, Yuqing, Awata, Junya, Parry, David A D, Marshall, Wallace F, Witman, George B, Lu, Xiangyi
• Format: Journal Article
• Language: English
• Published: United States The American Society for Cell Biology 01.04.2011
• Subjects: Animals; Calcium - metabolism; Cell Movement - physiology; Chlamydomonas reinhardtii - metabolism; Drosophila melanogaster - anatomy & histology; Drosophila melanogaster - metabolism; Drosophila Proteins - genetics; Drosophila Proteins - metabolism; Female; Flagella - physiology; Flagella - ultrastructure; Male; Microtubule-Associated Proteins - genetics; Microtubule-Associated Proteins - metabolism; Mutation; Signal Transduction - physiology; Spermatozoa - metabolism; TRPP Cation Channels - genetics; TRPP Cation Channels - metabolism
• ISSN: 1059-1524; 1939-4586
• DOI: 10.1091/mbc.e10-04-0331

Eukaryotic cilia and flagella are vital sensory and motile organelles. The calcium channel PKD2 mediates sensory perception on cilia and flagella, and defects in this can contribute to ciliopathic diseases. Signaling from Pkd2-dependent Ca²+ rise in the cilium to downstream effectors may require intermediary proteins that are largely unknown. To identify these proteins, we carried out genetic screens for mutations affecting Drosophila melanogaster sperm storage, a process mediated by Drosophila Pkd2. Here we show that a new mutation lost boys (lobo) encodes a conserved flagellar protein CG34110, which corresponds to vertebrate Ccdc135 (E = 6e-78) highly expressed in ciliated respiratory epithelia and sperm, and to FAP50 (E = 1e-28) in the Chlamydomonas reinhardtii flagellar proteome. CG34110 localizes along the fly sperm flagellum. FAP50 is tightly associated with the outer doublet microtubules of the axoneme and appears not to be a component of the central pair, radial spokes, dynein arms, or structures defined by the mbo waveform mutants. Phenotypic analyses indicate that both Pkd2 and lobo specifically affect sperm movement into the female storage receptacle. We hypothesize that the CG34110/Ccdc135/FAP50 family of conserved flagellar proteins functions within the axoneme to mediate Pkd2-dependent processes in the sperm flagellum and other motile cilia.