Mouse Usp9x/Fam (fat facets in mouse) and its Drosophila ortholog faf (fat facets) encode substrate-specific deubiquitylating enzymes and are essential for early embryonic development. The zebrafish (Danio rerio) is a powerful tool for studying embryonic gene expression patterns and function, and to that end, we sought to characterize the zebrafish Usp9 ortholog. Zebrafish usp9 was identified from database searches, and the predicted Usp9 protein is very highly conserved in mouse (90% identical and 94% similar) over its entire length. Phylogenetic analysis indicated that vertebrate Usp9s are highly clustered and separate from the USP9Y and Drosophila forms. We examined the developmental expression of usp9 from fertilization to 2 days postfertilization. usp9 is initially expressed ubiquitously but later restricted to the cephalic central nervous system, the developing lens, distal tips of the pectoral fin bud, and migrating endoderm. The extraordinary level of conservation between the mouse and zebrafish genes, coupled with equivalent expression patterns, makes zebrafish an appropriate complementary system for the study of usp9 in development.