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Elife 2020 Dec 02;9. doi: 10.7554/eLife.58662.
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Functional partitioning of a liquid-like organelle during assembly of axonemal dyneins.

Lee C , Cox RM , Papoulas O , Horani A , Drew K , Devitt CC , Brody SL , Marcotte EM , Wallingford JB .

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.

PubMed ID: 33263282
PMC ID: PMC7785291
Article link: Elife
Grant support: [+]

Species referenced: Xenopus laevis
Genes referenced: dnaaf11 dnaaf2 dnaaf5 dnaaf8 dnah10 dnah2 dnah9 dnai1 dnai2 dnai3 dnai4 dnai7 dnal1 dnal4 dnali1 dynll2 dynlrb1 dynlt1 dynlt2b hsp90ab1 lhx6 mcc mtor nme9 ruvbl1 ruvbl2 sf3a3 spag1 stip1 wdr18 zmynd10
Morpholinos: dnaaf8 MO1 dnaaf8 MO2

Disease Ontology terms: primary ciliary dyskinesia

Article Images: [+] show captions
References [+] :
Anderegg, NME5 frameshift variant in Alaskan Malamutes with primary ciliary dyskinesia. 2019, Pubmed