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Brain Res Dev Brain Res ; 150(2): 89-101, 2004 Jun 21.
Article in English | MEDLINE | ID: mdl-15158073

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.


Subject(s)
Caenorhabditis elegans Proteins/physiology , Embryonic and Fetal Development/physiology , Gene Expression Regulation, Developmental , Genotype , Microfilament Proteins/physiology , Sensation Disorders/genetics , Sequence Homology , Animals , Behavior, Animal , Blotting, Northern/methods , Caenorhabditis elegans Proteins/genetics , Cloning, Molecular , Embryo, Mammalian , Exploratory Behavior/physiology , Female , Humans , In Situ Hybridization/methods , Mice , Mice, Inbred Strains , Mice, Mutant Strains , Microfilament Proteins/genetics , Motor Activity/genetics , Mutation , Optic Disk/growth & development , Optic Disk/pathology , Optic Nerve/growth & development , Optic Nerve/pathology , Pain/genetics , Pain Measurement/methods , Pregnancy , Psychomotor Performance/physiology , RNA, Messenger/biosynthesis , Reaction Time/genetics , Reflex, Startle/genetics , Reverse Transcriptase Polymerase Chain Reaction/methods , Rotarod Performance Test/methods
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