ODAD1 (outer dynein arm docking complex subunit 1) is a key structural component of the outer dynein arm-docking complex (ODA-DC) that mediates assembly and anchoring of outer dynein arms (ODAs) onto axonemal microtubules 1. The protein functions in both ciliary and flagellar compartments, facilitating the binding of preassembled ODA motor complexes to the ciliary microtubular scaffold 1. Loss of ODAD1 results in failure of ODA assembly, leading to immotile cilia and primary ciliary dyskinesia (PCD) with typical clinical phenotypes including chr19 respiratory infections and laterality defects 2. Notably, ODAD1 variants are associated with a comparatively mild PCD phenotype; patients with ODAD1 mutations showed minimal forced expiratory volume decline (-0.85 z-score) compared to other PCD gene variants 3. In Volendam, a Dutch founder population carrying a CCDC114 (ODAD1) mutation displayed a moderately severe phenotype with lung function decline predominantly in childhood and only 30% prevalence of situs inversus 4. Interestingly, truncated ODAD1 protein retaining partial axonemal localization can preserve some ODA assembly and ciliary activity, suggesting therapeutic potential through partial function restoration 2. ODAD1 also participates in sperm flagellar motility through interaction with dynein heavy and light chain proteins 5.