title: Syntrophin-2 is required for eye development in Drosophila.
abstract: Syntrophins are components of the dystrophin glycoprotein complex (DGC), which is encoded by causative genes of muscular dystrophies. The DGC is thought to play roles not only in linking the actin cytoskeleton to the extracellular matrix, providing stability to the cell membrane, but also in signal transduction. Because of their binding to a variety of different molecules, it has been suggested that syntrophins are adaptor proteins recruiting signaling proteins to membranes and the DGC. However, critical roles in vivo remain elusive. Drosophila Syntrophin-2 (Syn2) is an orthologue of human gamma 1/gamma 2-syntrophins. Western immunoblot analysis here showed Syn2 to be expressed throughout development, with especially high levels in the adult head. Morphological aberrations were observed in Syn2 knockdown adult flies, with lack of retinal elongation and malformation of rhabdomeres. Furthermore, Syn2 knockdown flies exhibited excessive apoptosis in third instar larvae and alterations in the actin localization in the pupal retinae. Genetic crosses with a collection of Drosophila deficiency stocks allowed us to identify seven genomic regions, deletions of which caused enhancement of the rough eye phenotype induced by Syn2 knockdown. This information should facilitate identification of Syn2 regulators in Drosophila and clarification of roles of Syn2 in eye development.
authors.affiliation: Department of Applied Biology, Kyoto Institute of Technology, Matsugasaki, Sakyo-ku, Kyoto 606-8585, Japan.
authors.forename: Rika
authors.initials: R
authors.lastname: Nagai
authors.forename: Reina
authors.initials: R
authors.lastname: Hashimoto
authors.forename: Yuko
authors.initials: Y
authors.lastname: Tanaka
authors.forename: Osamu
authors.initials: O
authors.lastname: Taguchi
authors.forename: Mamiko
authors.initials: M
authors.lastname: Sato
authors.forename: Akio
authors.initials: A
authors.lastname: Matsukage
authors.forename: Masamitsu
authors.initials: M
authors.lastname: Yamaguchi
mesh_terms: D000199:Actins / Q000378:metabolism; D048868:Adaptor Proteins, Signal Transducing / Q000235:genetics; D000595:Amino Acid Sequence; D000818:Animals; D000918:Antibody Specificity / Q000276:immunology; D017209:Apoptosis / Q000235:genetics; D051017:Apoptosis Regulatory Proteins / Q000235:genetics; D001921:Brain / Q000378:metabolism; D053148:Caspase 3 / Q000378:metabolism; D004330:Drosophila / Q000235:genetics / Q000254:growth & development* / Q000378:metabolism*; D029721:Drosophila Proteins / Q000502:physiology*; D049029:Dystrophin-Associated Proteins / Q000502:physiology*; D004625:Embryo, Nonmammalian / Q000378:metabolism; D005123:Eye / Q000254:growth & development* / Q000378:metabolism* / Q000473:pathology / Q000648:ultrastructure; D005260:Female; D017353:Gene Deletion; D006801:Humans; D007814:Larva / Q000378:metabolism; D008297:Male; D008565:Membrane Proteins / Q000235:genetics; D008969:Molecular Sequence Data; D009024:Morphogenesis / Q000502:physiology*; D009124:Muscle Proteins / Q000235:genetics; D017956:Photoreceptor Cells, Invertebrate / Q000378:metabolism / Q000473:pathology; D011679:Pupa / Q000378:metabolism; D012160:Retina / Q000254:growth & development / Q000473:pathology; D017386:Sequence Homology, Amino Acid
chemical_list: D000199:Actins; D048868:Adaptor Proteins, Signal Transducing; D051017:Apoptosis Regulatory Proteins; D029721:Drosophila Proteins; D049029:Dystrophin-Associated Proteins; D008565:Membrane Proteins; D009124:Muscle Proteins; C432243:SNTG1 protein, human; C410037:SNTG2 protein, human; C546324:Syntrophin-2, Drosophila; C091824:syntrophin; D053148:Caspase 3