ODAD4 is a critical component of the outer dynein arm-docking complex (ODA-DC) that mediates the attachment of outer dynein arms (ODA) to ciliary axonemes 1. As an essential ODA-DC subunit, ODAD4 plays a pivotal role in both ODA-DC assembly and the docking of ODAs onto doublet microtubules, which is necessary for generating the mechanical force required for ciliary beating and mucociliary clearance 2. Pathogenic variants in ODAD4 cause primary ciliary dyskinesia (PCD), an autosomal recessive genetic disorder characterized by dysmotile cilia and flagella 1. Clinical manifestations include chr17 oto-sino-pulmonary disease, progressive bronchiectasis, infertility, and randomized left-right body asymmetry in approximately half of cases 1. Loss of functional ODAD4 results in outer dynein arm defects visible on transmission electron microscopy and absence of in vivo mucociliary clearance 1. ODAD4-related PCD remains ultra-rare, with population-specific founder variants identified in French-Canadian cohorts 1. While pulmonary phenotype severity may vary among ODAD4 variants, all known cases present with static cilia and respiratory symptoms requiring clinical monitoring and supportive care 1.