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TitleSystematic studies of all PIH proteins in zebrafish reveal their distinct roles in axonemal dynein assembly.
Journal, issue, pagesElife, Vol. 7, Year 2018
Publish dateMay 9, 2018
AuthorsHiroshi Yamaguchi / Toshiyuki Oda / Masahide Kikkawa / Hiroyuki Takeda /
PubMed AbstractConstruction of motile cilia/flagella requires cytoplasmic preassembly of axonemal dyneins before transport into cilia. Axonemal dyneins have various subtypes, but the roles of each dynein subtype ...Construction of motile cilia/flagella requires cytoplasmic preassembly of axonemal dyneins before transport into cilia. Axonemal dyneins have various subtypes, but the roles of each dynein subtype and their assembly processes remain elusive in vertebrates. The PIH protein family, consisting of four members, has been implicated in the assembly of different dynein subtypes, although evidence for this idea is sparse. Here, we established zebrafish mutants of all four PIH-protein genes: , , , and , and analyzed the structures of axonemal dyneins in mutant spermatozoa by cryo-electron tomography. Mutations caused the loss of specific dynein subtypes, which was correlated with abnormal sperm motility. We also found organ-specific compositions of dynein subtypes, which could explain the severe motility defects of mutant Kupffer's vesicle cilia. Our data demonstrate that all vertebrate PIH proteins are differently required for cilia/flagella motions and the assembly of axonemal dyneins, assigning specific dynein subtypes to each PIH protein.
External linksElife / PubMed:29741156 / PubMed Central
MethodsEM (tomography)
Resolution41.7 - 45.1 Å
Structure data

EMDB-6954:
Doublet microtubule of zebrafish sperm axoneme, WT
Method: EM (tomography) / Resolution: 42.5 Å

EMDB-6955:
Doublet microtubule of zebrafish sperm axoneme, pih1d1_null mutant
Method: EM (tomography) / Resolution: 45.0 Å

EMDB-6956:
Doublet microtubule of zebrafish sperm axoneme, pih1d2_null mutant
Method: EM (tomography) / Resolution: 45.1 Å

EMDB-6957:
Doublet microtubule of zebrafish sperm axoneme, ktu_null mutant
Method: EM (tomography) / Resolution: 43.4 Å

EMDB-6958:
Doublet microtubule of zebrafish sperm axoneme, twister_null mutant
Method: EM (tomography) / Resolution: 43.7 Å

EMDB-6959:
Doublet microtubule of zebrafish sperm axoneme, pih1d2_null and ktu_null double_mutant, +OAD
Method: EM (tomography) / Resolution: 43.4 Å

EMDB-6960:
Doublet microtubule of zebrafish sperm axoneme, pih1d2_null and ktu_null double_mutant, -OAD
Method: EM (tomography) / Resolution: 41.7 Å

Source
  • Danio rerio (zebrafish)

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