Our initial description of MNS1 as a cause of laterality defects and male infertility in humans included a particularly revealing finding, whereby ciliary ultrastructural analyses from the female with biallelic pathogenic variants in MNS1 and DNAH5 shed light on MNS1’s specific involvement in the docking process of the ODA (ODA-DC) at the distal ends of respiratory axonemes. The gene discussed is DNAH5; the disease is male infertility.