The observation that axonemes of spermatozoa were abnormal in groups of patients with X-Linked RP [30] and Usher syndrome [31] was made more than 20 years ago before the identification of ciliary genes, such as retinitis pigmentosa type 1(RP1), retinitis pigmentosa GTPase regulator gene (RPGR), retinitis pigmentosa GTPase regulator-interacting protein (RPGRIP1), and the Usher syndrome genes [32]. The gene discussed is RPGRIP1; the disease is Usher syndrome.