title: Proviral insertions in the zebrafish hagoromo gene, encoding an F-box/WD40-repeat protein, cause stripe pattern anomalies.
abstract: The zebrafish, Danio rerio, has three types of pigment cells (melanophores, xanthophores and iridophores) and, in adult fish, these cells are organized into a stripe pattern. The mechanisms underlying formation of the stripe pattern are largely unknown. We report here the identification and characterization of a novel dominant zebrafish mutation, hagoromo (hag), which was generated by insertional mutagenesis using a pseudotyped retrovirus. The hag mutation caused disorganized stripe patterns. Two hag mutant alleles were isolated independently and proviruses were located within the fifth intron of a novel gene, which we named hag, encoding an F-box/WD40-repeat protein. The hag gene was mapped to linkage group (LG)13, close to fgf8 and pax2.1. Amino acid sequence similarity, conserved exon-intron boundaries and conserved synteny indicated that zebrafish hag is an ortholog of mouse Dactylin, the gene mutated in the Dactylaplasia (Dac) mouse [1]. The Dac mutation is dominant and causes defects in digit formation in fore- and hindlimbs. This study revealed that the hag locus is important for pattern formation in fish but is involved in distinct morphogenetic events in different vertebrates.
authors.affiliation: Department of Tumor Biology, The Institute of Medical Science, The University of Tokyo, Minato-ku, 108-8639, Japan. kawakami@ims.u-tokyo.ac.jp
authors.forename: K
authors.initials: K
authors.lastname: Kawakami
authors.forename: A
authors.initials: A
authors.lastname: Amsterdam
authors.forename: N
authors.initials: N
authors.lastname: Shimoda
authors.forename: T
authors.initials: T
authors.lastname: Becker
authors.forename: J
authors.initials: J
authors.lastname: Mugg
authors.forename: A
authors.initials: A
authors.lastname: Shima
authors.forename: N
authors.initials: N
authors.lastname: Hopkins
mesh_terms: D000595:Amino Acid Sequence; D000818:Animals; D019521:Body Patterning / Q000235:genetics*; D018076:DNA, Complementary / Q000235:genetics; D044783:F-Box Proteins; D051379:Mice; D008969:Molecular Sequence Data; D016254:Mutagenesis, Insertional; D011506:Proteins / Q000235:genetics*; D011533:Proviruses / Q000235:genetics; D012190:Retroviridae / Q000235:genetics; D016415:Sequence Alignment; D015027:Zebrafish / Q000235:genetics*; D029961:Zebrafish Proteins
chemical_list: D018076:DNA, Complementary; D044783:F-Box Proteins; C120449:FBXW4 protein, human; C501124:Fbxw4 protein, mouse; D011506:Proteins; D029961:Zebrafish Proteins; C501123:fbxw4 protein, zebrafish