DNAAF1 (dynein axonemal assembly factor 1) is a cytoplasmic protein essential for ciliary function and cardiac development. Functionally, DNAAF1 is required for dynein heavy chain assembly and the stability of ciliary architecture, playing a central role in cytoplasmic preassembly of dynein-arm complexes 1. The protein interacts with ciliary intraflagellar transport proteins and AAA+ ATPases (RUVBL1 and RUVBL2), interactions critical for proper cilia motility and asymmetric left-right patterning during development 1. Disease relevance is substantial. DNAAF1 mutations cause primary ciliary dyskinesia (PCD), characterized by defective ciliary motility, infertility, bronchiectasis, and sinusitis 23. Beyond PCD, DNAAF1 mutations are associated with heterotaxy and congenital heart defects 14, neural tube defects 5, adolescent idiopathic scoliosis 6, and testicular germ cell tumors 7, suggesting broader roles in developmental morphogenesis and cancer susceptibility. Clinically, whole-exome sequencing identifies DNAAF1 mutations in ~56% of laterality disorder cases 4. Gene therapy shows promise: lentiviral vectors successfully restore DNAAF1 expression and ciliary function in patient-derived airway organoids 8, providing proof-of-concept for targeted therapeutic approaches.