title: Regulation of flagellar motility by the conserved flagellar protein CG34110/Ccdc135/FAP50.
abstract: 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.
authors.affiliation: Institute of Environmental Health Sciences and Department of Biochemistry and Molecular Biology, Wayne State University, Detroit, MI 48201, USA.
authors.forename: Yong
authors.initials: Y
authors.lastname: Yang
authors.forename: Deborah A
authors.initials: DA
authors.lastname: Cochran
authors.forename: Mary D
authors.initials: MD
authors.lastname: Gargano
authors.forename: Iryna
authors.initials: I
authors.lastname: King
authors.forename: Nayef K
authors.initials: NK
authors.lastname: Samhat
authors.forename: Benjain P
authors.initials: BP
authors.lastname: Burger
authors.forename: Katherine R
authors.initials: KR
authors.lastname: Sabourin
authors.forename: Yuqing
authors.initials: Y
authors.lastname: Hou
authors.forename: Junya
authors.initials: J
authors.lastname: Awata
authors.forename: David A D
authors.initials: DA
authors.lastname: Parry
authors.forename: Wallace F
authors.initials: WF
authors.lastname: Marshall
authors.forename: George B
authors.initials: GB
authors.lastname: Witman
authors.forename: Xiangyi
authors.initials: X
authors.lastname: Lu
mesh_terms: D000818:Animals; D002118:Calcium / Q000378:metabolism; D002465:Cell Movement / Q000502:physiology*; D016825:Chlamydomonas reinhardtii / Q000378:metabolism; D029721:Drosophila Proteins / Q000235:genetics / Q000378:metabolism*; D004331:Drosophila melanogaster / Q000033:anatomy & histology* / Q000378:metabolism; D005260:Female; D005407:Flagella / Q000502:physiology* / Q000648:ultrastructure; D008297:Male; D008869:Microtubule-Associated Proteins / Q000235:genetics / Q000378:metabolism*; D009154:Mutation; D015398:Signal Transduction / Q000502:physiology; D013094:Spermatozoa / Q000378:metabolism; D050396:TRPP Cation Channels / Q000235:genetics / Q000378:metabolism
chemical_list: D002118:Calcium; D029721:Drosophila Proteins; D008869:Microtubule-Associated Proteins; D050396:TRPP Cation Channels; C568284:lobo protein, Drosophila