Annotation inferences using phylogenetic trees
Gene Ontology annotation based on UniProtKB/Swiss-Prot Subcellular Location vocabulary mapping, accompanied by conservative changes to GO terms applied by UniProt
Automatic assignment of GO terms using logical inference, based on on inter-ontology links
LRRC50, a conserved ciliary protein implicated in polycystic kidney disease.
Deletions and point mutations of LRRC50 cause primary ciliary dyskinesia due to dynein arm defects.
Loss-of-function mutations in the human ortholog of Chlamydomonas reinhardtii ODA7 disrupt dynein arm assembly and cause primary ciliary dyskinesia.
DNAAF1 links heart laterality with the AAA+ ATPase RUVBL1 and ciliary intraflagellar transport.
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Human DNAAF1 binds IFT88 in a one-to-one yeast two-hybrid assay and co-immunoprecipitates with endogenous or expressed IFT88 in HEK293 cells; the IFT88 C-terminal region supports the Y2H interaction.
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Human DNAAF1 co-purifies with RUVBL1/RUVBL2, with endogenous-partner co-immunoprecipitation confirming association. RUVBL1 depletion perturbs DNAAF1/IFT88 colocalization; the assembly-to-transport handoff remains a proposed model.
ZMYND10 stabilizes intermediate chain proteins in the cytoplasmic pre-assembly of dynein arms.
Targeted deletion of the AAA-ATPase Ruvbl1 in mice disrupts ciliary integrity and causes renal disease and hydrocephalus.