Figure 2.

Positional cloning and characterization of fleer mRNAs. (A) flr is located on chromosome 3, tightly linked to flanking SSR markers z10805 (1/578 meioses) and z63912 (1/1450 meioses). BAC zC76L23, located within the flr interval, contained a predicted TPR protein that is present in multiple cilia proteome databases and thus a potential flr candidate. (B) cDNA sequence of the flr candidate from WT and flr−/− embryos identified a T (798) > A nonsense mutation (arrowhead) that replaces a wild-type tyrosine (266) with an ochre stop codon. (C) The flr gene contains 19 exons which encode two mRNA isoforms. In flr_tv1, an upstream splice donor is used in exon 12 and exon 17 is omitted. (D) Fleer and Fleer_tv1 are predicted to contain four TPR motifs and a coiled-coil domain (CC). (E) Alignment of predicted Fleer related proteins from invertebrate and vertebrate species demonstrate evolutionary conservation. Red indicates identity, and yellow shading outlines conserved TPR domains.