biostudies-literatureYamaguchi HCore Research for Evolutional Science and TechnologyJapan Society for the Promotion of Sciencee36979https://www.ebi.ac.uk/biostudies/studies/S-EPMC6008050biostudies-literatureUnknown7Construction 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: <i>pih1d1</i>, <i>pih1d2</i>, <i>ktu</i>, and <i>twister</i>, 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 eLifeSystematic studies of all PIH proteins in zebrafish reveal their distinct roles in axonemal dynein assembly.PMC600805016H02502JPMJCR14M1Oda TYamaguchi HKikkawa MTakeda HfalseSystematic studies of all PIH proteins in zebrafish reveal their distinct roles in axonemal dynein assembly.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: <i>pih1d1</i>, <i>pih1d2</i>, <i>ktu</i>, and <i>twister</i>, 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 2018-01-01T00:00:00Z2018 May2026-04-29T17:45:26.275Z2019-03-26T23:42:33ZS-EPMC60080502974115610.7554/eLife.36979