Fig. 7

swt encodes lrrc50, a novel gene required for cilia motility. (A) Schematic diagram of the LRRC50 protein highlighting the location of tm317 and fk03a mutations. LRRC50 contains 6 predicted leucine rich repeats (cylinders), followed by a coiled-coil domain (spiral); however, the functions of these motifs in Swt are unknown. (B) Sequence traces of wild-type sibling and mutant DNA, showing the mutations in each allele. The swt^tm317 allele creates a missense mutation changing AAC (ASN) to AAA (LYS) at amino acid position 172 in the leucine rich repeat region. The swt^fk03a allele creates a nonsense mutation changing GAG (GLU) to TAG (stop codon) at amino acid position 259 after the leucine rich repeats before the coiled-coil domain. swt RNA is expressed in cells that contain cilia: (C) Kupffer′s vesicle (arrow); (D) neural tube staining (arrowhead) and bilateral intermediate mesoderm staining (arrows); (E) otic vesicle (arrow); (F) cross-section showing expression i cells that contain cilia: (C) Kupffer′s vesicle (arrow); (D) neural tube staining (arrowhead) and bilateral intermediate mesodn the floorplate of the neural tube (arrowhead); (G) lateral view of otic vesicle expression (arrow); (H) pronephric duct (arrow, dorsal to the yolk extension) and chordo-neural hinge expression (arrowhead, at the tip of the tail). Embryo in panel C is at tailbud stage (dorsal view of posterior), panels D–G are at the 12 somite stage, and panel H is at 24 hpf. In panels D and E, anterior is up.