DNAAF11 (LRRC6) is a cytoplasmic protein essential for dynein arm assembly in motile cilia and flagella 1. The protein is involved in the assembly and transport of both inner and outer dynein arms, which are crucial for proper ciliary motility 1 2. DNAAF11 interacts with ZMYND10, forming a cytoplasmic protein complex necessary for motile ciliary function 2. During ciliogenesis, DNAAF11 is upregulated in human airway epithelial cells in a Foxj1-dependent manner and localizes to the cytoplasm 1. Biallelic mutations in DNAAF11 cause primary ciliary dyskinesia (PCD), a genetic disorder characterized by recurrent respiratory infections, male infertility, and laterality defects 1 3. In PCD patients with DNAAF11 mutations, dynein arm proteins are either absent or mislocalized to the cytoplasm, resulting in absent dynein arms and reduced cilia beat frequency 1. Male patients with DNAAF11 mutations exhibit sperm with aberrant morphology and ultrastructure lacking inner and outer dynein arms, leading to infertility that can be treated with intracytoplasmic sperm injection 3. DNAAF11 mutations have been identified in multiple populations, including Palestinian, Indian, Vietnamese, and Chinese patients with PCD 4 5 6 3.