DNAH14 encodes dynein axonemal heavy chain 14, a force-generating protein essential for respiratory cilia function and sperm motility. The protein produces force towards microtubule minus ends through ATPase activity, with the power stroke occurring upon ADP release [UniProt]. DNAH14 functions in cilium movement and microtubule motor activity within dynein complexes [GO Annotations]. Recent evidence has expanded DNAH14's clinical significance beyond ciliary disorders to include neurodevelopmental conditions. Pathogenic variants in DNAH14 have been associated with primary ciliary dyskinesia (PCD), a heterogeneous disorder affecting respiratory cilia function 1 2. Additionally, compound heterozygous DNAH14 variants cause previously unrecognized neurodevelopmental disorders characterized by seizures, global developmental delay, microcephaly, and hypotonia, with variants typically affecting the conserved AAA+ ATPase domain 3. DNAH14 variants have also been identified in intellectual disability cases from consanguineous populations 4. Furthermore, genetic variations in DNAH14 may serve as biomarkers for treatment response in cystic fibrosis lung disease 5 and neoadjuvant chemoradiotherapy response in rectal cancer 6. These findings suggest DNAH14 plays broader roles beyond ciliary function, potentially affecting neurological development and disease progression.