DNAL4 (dynein axonemal light chain 4) is a force-generating protein component of respiratory cilia that produces force toward the minus ends of microtubules through ATPase activity 1. The gene encodes a dynein light chain involved in microtubule-based motor activity and protein binding functions. DNAL4 has been identified as a genetic cause of congenital mirror movements (CMM), a rare neurodevelopmental disorder characterized by involuntary movements mirroring voluntary movements on the opposite body side 12. A homozygous splice site mutation in DNAL4 was identified in a large consanguineous Pakistani family with autosomal recessive CMM inheritance, resulting in exon 3 skipping and omission of 28 amino acids 1. DNAL4 mutations account for a minority of CMM cases; among 80 affected individuals, pathogenic variants were identified in only a subset, with DNAL4 being one of five known CMM-associated genes alongside DCC, RAD51, NTN1, and ARHGEF7 2. The mechanism by which DNAL4 dysfunction causes CMM may involve impaired cytoplasmic dynein complex function required for netrin-1-directed retrograde transport in commissural neurons of the corpus callosum, affecting cross-brain wiring 1. Unlike typical axonemal dyneins, DNAL4-associated CMM does not present with primary ciliary dyskinesis or situs inversus, suggesting a specific neurological role distinct from conventional ciliary function.