DRC2 (dynein regulatory complex subunit 2), also known as CCDC65, is a critical structural component of the nexin-dynein regulatory complex (N-DRC), which controls ciliary and flagellar motility 1. DRC2 functions as a central hub for N-DRC assembly, with its C-terminal region essential for coassembling with DRC1 to form the base plate that attaches to outer doublet microtubules 1. This assembly stabilizes multiple associated axonemal structures, including inner dynein arms, radial spokes, and the calmodulin-spoke complex 1. Mutations in DRC2/CCDC65 cause primary ciliary dyskinesia (PCD), a genetic ciliopathy characterized by impaired ciliary function and dyskinetic cilia beating 2. Notably, DRC2 mutations can produce PCD without detectable ultrastructural ciliary abnormalities 2, highlighting the functional role of N-DRC components. Loss of DRC2 disrupts assembly of multiple N-DRC subunits and destabilizes associated dynein arms 1. Beyond respiratory cilia, DRC2 is essential for sperm flagellum structure and function 3. Mutations in CCDC65 cause multiple morphological abnormalities of sperm flagella (MMAF) with asthenozoospermia, demonstrating that DRC2 function extends to male reproductive biology 3. These findings establish DRC2 as a fundamental regulator of axonemal dynamics with implications for both respiratory ciliopathies and male infertility.