“…That the sensory mechanism localizes to the cilium is supported by the fact that mutations in genes that affect JO ciliary development and morphology lead to deafness. These include components of intraflagellar transport (IFT) such as the anterograde kinesin motor subunits kinesin-like protein 64D (Klp64D) and the kinesin associated protein (DmKAP) (Sarpal et al, 2003), the retrograde cytoplasmic dynein motor encoded by beethoven (btv) (Eberl et al, 2000, Sharma et al, in preparation), the anterograde IFT-B particle proteins no mechanoreceptor potential B (nompB) (Han et al, 2003), outer segment protein 2 (oseg2; also called osm-1) and outer segment protein 5 (oseg5) (Avidor-Reiss et al, 2004), the retrograde IFT-A particle proteins oseg1 and reduced mechanoreceptor potential A (rempA; also called oseg3) (Avidor-Reiss et al, 2004, Lee et al, in preparation) and the ciliary transcription regulator Drosophila regulatory factor X (dRfx) (Durand et al, 2000, Dubruille et al, 2002. Mutations that appear to affect the ciliary axoneme also lead to deafness.…”