A role for the C. elegans L1CAM homologue lad-1/sax-7 in maintaining tissue attachment.
The L1 family of cell adhesion molecules (L1CAMs) is important for neural development. Mutations in one of the human L1CAM genes, L1, can result in several neurological syndromes, the symptoms of which are variably penetrant. The physiological cause of these symptoms, collectively termed CRASH, is not clear. Caenorhabditis elegans animals genetically null for the L1CAM homologue LAD-1, exhibit variably penetrant pleiotropic phenotypes that are similar to the CRASH symptoms; thus the C. elegans lad-1 mutant provides an excellent model system to study how disruption of L1 leads to these abnormalities. These phenotypes include uncoordinated movements, variable embryonic lethality, and abnormal neuronal distribution and axon trajectories. Our analysis revealed that many of these phenotypes are likely a result of tissue detachment.
Wang
Xuelin
X
Department of Genetics, Cell Biology, and Development, The Developmental Biology Center, University of Minnesota, 6-160 Jackson Hall, SE Minneapolis, MN 55455, USA.
Kweon
Junghun
J
Larson
Stephanie
S
Chen
Lihsia
L
D000818:Animals; D030801:Animals, Genetically Modified; D001369:Axons; D017173:Caenorhabditis elegans / Q000166:cytology / Q000196:embryology / Q000235:genetics / Q000378:metabolism*; D029742:Caenorhabditis elegans Proteins / Q000235:genetics / Q000378:metabolism*; D002448:Cell Adhesion; D015816:Cell Adhesion Molecules, Neuronal / Q000235:genetics / Q000378:metabolism*; D002874:Chromosome Mapping; D017238:Genes, Helminth; D017930:Genes, Reporter; D005816:Genetic Complementation Test; D049452:Green Fluorescent Proteins / Q000378:metabolism; D015801:Helminth Proteins / Q000235:genetics / Q000378:metabolism*; D018715:Microscopy, Video; D008954:Models, Biological; D009024:Morphogenesis; D009132:Muscles / Q000196:embryology / Q000378:metabolism; D009154:Mutation; D039842:Neural Cell Adhesion Molecule L1 / Q000235:genetics / Q000378:metabolism*; D019006:Neural Cell Adhesion Molecules / Q000235:genetics / Q000378:metabolism*; D009474:Neurons / Q000166:cytology / Q000378:metabolism; D020033:Protein Isoforms / Q000235:genetics / Q000378:metabolism
D029742:Caenorhabditis elegans Proteins; D015816:Cell Adhesion Molecules, Neuronal; D015801:Helminth Proteins; D039842:Neural Cell Adhesion Molecule L1; D019006:Neural Cell Adhesion Molecules; D020033:Protein Isoforms; C501528:SAX-7 protein, C elegans; D049452:Green Fluorescent Proteins