title: Pih1d3 is required for cytoplasmic preassembly of axonemal dynein in mouse sperm.
abstract: Axonemal dynein complexes are preassembled in the cytoplasm before their transport to cilia, but the mechanism of this process remains unclear. We now show that mice lacking Pih1d3, a PIH1 domain-containing protein, develop normally but manifest male sterility. Pih1d3(-/-) sperm were immotile and fragile, with the axoneme of the flagellum lacking outer dynein arms (ODAs) and inner dynein arms (IDAs) and showing a disturbed 9+2 microtubule organization. Pih1d3 was expressed specifically in spermatogenic cells, with the mRNA being most abundant in pachytene spermatocytes. Pih1d3 localized to the cytoplasm of spermatogenic cells but was not detected in spermatids or mature sperm. The levels of ODA and IDA proteins were reduced in the mutant testis and sperm, and Pih1d3 was found to interact with an intermediate chain of ODA as well as with Hsp70 and Hsp90. Our results suggest that Pih1d3 contributes to cytoplasmic preassembly of dynein complexes in spermatogenic cells by stabilizing and promoting complex formation by ODA and IDA proteins.
authors.affiliation: Developmental Genetics Group, Graduate School of Frontier Biosciences, Osaka University, and Core Research for Evolutional Science and Technology (CREST), Japan Science and Technology Corporation (JST), 1-3 Yamada-oka, Suita, Osaka 565-0871, Japan.
authors.forename: Fenglan
authors.initials: F
authors.lastname: Dong
authors.forename: Kyosuke
authors.initials: K
authors.lastname: Shinohara
authors.forename: Yanick
authors.initials: Y
authors.lastname: Botilde
authors.forename: Ryo
authors.initials: R
authors.lastname: Nabeshima
authors.forename: Yasuko
authors.initials: Y
authors.lastname: Asai
authors.forename: Akemi
authors.initials: A
authors.lastname: Fukumoto
authors.forename: Toshiaki
authors.initials: T
authors.lastname: Hasegawa
authors.forename: Moe
authors.initials: M
authors.lastname: Matsuo
authors.forename: Hiroyuki
authors.initials: H
authors.lastname: Takeda
authors.forename: Hidetaka
authors.initials: H
authors.lastname: Shiratori
authors.forename: Tetsuya
authors.initials: T
authors.lastname: Nakamura
authors.forename: Hiroshi
authors.initials: H
authors.lastname: Hamada
mesh_terms: D000818:Animals; D051017:Apoptosis Regulatory Proteins / Q000235:genetics / Q000378:metabolism / Q000502:physiology*; D056444:Axonemal Dyneins / Q000378:metabolism*; D054468:Axoneme / Q000378:metabolism; D003593:Cytoplasm / Q000378:metabolism / Q000648:ultrastructure; D005298:Fertility / Q000235:genetics; D018840:HSP70 Heat-Shock Proteins / Q000378:metabolism; D018841:HSP90 Heat-Shock Proteins / Q000378:metabolism; D047908:Intracellular Signaling Peptides and Proteins; D008297:Male; D051379:Mice; D012333:RNA, Messenger / Q000378:metabolism; D013094:Spermatozoa / Q000378:metabolism* / Q000648:ultrastructure; D013737:Testis / Q000378:metabolism
chemical_list: D051017:Apoptosis Regulatory Proteins; D018840:HSP70 Heat-Shock Proteins; D018841:HSP90 Heat-Shock Proteins; C490434:Hspa2 protein, mouse; D047908:Intracellular Signaling Peptides and Proteins; C587686:PIH1D3 protein, mouse; D012333:RNA, Messenger; D056444:Axonemal Dyneins