TY - JOUR TI - Functional partitioning of a liquid-like organelle during assembly of axonemal dyneins AU - Lee, Chanjae AU - Cox, Rachael M AU - Papoulas, Ophelia AU - Horani, Amjad AU - Drew, Kevin AU - Devitt, Caitlin C AU - Brody, Steven L AU - Marcotte, Edward M AU - Wallingford, John B A2 - Carter, Andrew P A2 - Akhmanova, Anna A2 - Stearns, Tim A2 - Kikkawa, Masahide VL - 9 PY - 2020 DA - 2020/12/02 SP - e58662 C1 - eLife 2020;9:e58662 DO - 10.7554/eLife.58662 UR - https://doi.org/10.7554/eLife.58662 AB - Ciliary motility is driven by axonemal dyneins that are assembled in the cytoplasm before deployment to cilia. Motile ciliopathy can result from defects in the dyneins themselves or from defects in factors required for their cytoplasmic pre-assembly. Recent work demonstrates that axonemal dyneins, their specific assembly factors, and broadly-acting chaperones are concentrated in liquid-like organelles in the cytoplasm called DynAPs (Dynein Axonemal Particles). Here, we use in vivo imaging in Xenopus to show that inner dynein arm (IDA) and outer dynein arm (ODA) subunits are partitioned into non-overlapping sub-regions within DynAPs. Using affinity- purification mass-spectrometry of in vivo interaction partners, we also identify novel partners for inner and outer dynein arms. Among these, we identify C16orf71/Daap1 as a novel axonemal dynein regulator. Daap1 interacts with ODA subunits, localizes specifically to the cytoplasm, is enriched in DynAPs, and is required for the deployment of ODAs to axonemes. Our work reveals a new complexity in the structure and function of a cell-type specific liquid-like organelle that is directly relevant to human genetic disease. KW - cilia KW - axonemal dyneins KW - DynAPs KW - liquid-like organelle KW - ciliopathy KW - primary ciliary dykinesia JF - eLife SN - 2050-084X PB - eLife Sciences Publications, Ltd ER -