title: Sensory deficits in mice hypomorphic for a mammalian homologue of unc-53.
abstract: The migration of cells and the extension of cellular processes along pathways to their defined destinations are crucial in the development of higher organisms. Caenorhabditis elegans unc-53 plays an important role in cell migration and the outgrowth of cellular processes such as axons. To gain further insight into the biological function of unc53H2, a recently identified mammalian homologue of unc-53, we have generated mice carrying a mutation of unc53H2 and provide evidence that unc53H2 is involved in neuronal development and, more specifically, the development of different sensory systems. The unc53H2 hypomorphic mouse showed a general impaired acuity of several sensory systems (olfactory, auditory, visual and pain sensation) which in case of the visual system was corroborated by the morphological observation of hypoplasia of the optic nerve. We hypothesize that in analogy with its C. elegans homologue, unc53H2 may play a role in the processes of cellular outgrowth and migration.
authors.affiliation: Johnson & Johnson Pharmaceutical Research and Development, A Division of Janssen Pharmaceutica N.V., Turnhoutseweg 30, B-2340 Beerse, Belgium.
authors.forename: Pieter J
authors.initials: PJ
authors.lastname: Peeters
authors.forename: Alexis
authors.initials: A
authors.lastname: Baker
authors.forename: Ilse
authors.initials: I
authors.lastname: Goris
authors.forename: Gie
authors.initials: G
authors.lastname: Daneels
authors.forename: Peter
authors.initials: P
authors.lastname: Verhasselt
authors.forename: Walter H M L
authors.initials: WH
authors.lastname: Luyten
authors.forename: Johan J G H
authors.initials: JJ
authors.lastname: Geysen
authors.forename: Stefan U
authors.initials: SU
authors.lastname: Kass
authors.forename: Dieder W E
authors.initials: DW
authors.lastname: Moechars
mesh_terms: D000818:Animals; D001522:Behavior, Animal; D015152:Blotting, Northern / Q000379:methods; D029742:Caenorhabditis elegans Proteins / Q000235:genetics / Q000502:physiology*; D003001:Cloning, Molecular; D004622:Embryo, Mammalian; D005314:Embryonic and Fetal Development / Q000502:physiology*; D005106:Exploratory Behavior / Q000502:physiology; D005260:Female; D018507:Gene Expression Regulation, Developmental; D005838:Genotype; D006801:Humans; D017403:In Situ Hybridization / Q000379:methods; D051379:Mice; D008815:Mice, Inbred Strains; D008817:Mice, Mutant Strains; D008840:Microfilament Proteins / Q000235:genetics / Q000502:physiology*; D009043:Motor Activity / Q000235:genetics; D009154:Mutation; D009898:Optic Disk / Q000254:growth & development / Q000473:pathology; D009900:Optic Nerve / Q000254:growth & development / Q000473:pathology; D010146:Pain / Q000235:genetics; D010147:Pain Measurement / Q000379:methods; D011247:Pregnancy; D011597:Psychomotor Performance / Q000502:physiology; D012333:RNA, Messenger / Q000096:biosynthesis; D011930:Reaction Time / Q000235:genetics; D013216:Reflex, Startle / Q000235:genetics; D020133:Reverse Transcriptase Polymerase Chain Reaction / Q000379:methods; D045442:Rotarod Performance Test / Q000379:methods; D012678:Sensation Disorders / Q000235:genetics*; D017385:Sequence Homology
chemical_list: D029742:Caenorhabditis elegans Proteins; D008840:Microfilament Proteins; D012333:RNA, Messenger; C465016:UNC-53 protein, C elegans