A role for the C. elegans L1CAM homologue lad-1/sax-7 in maintaining tissue attachment

Xuelin Wang, Junghun Kweon, Stephanie Larson, Lihsia Chen

Research output: Contribution to journalArticlepeer-review

37 Scopus citations

Abstract

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.

Original languageEnglish (US)
Pages (from-to)273-291
Number of pages19
JournalDevelopmental Biology
Volume284
Issue number2
DOIs
StatePublished - Aug 15 2005

Bibliographical note

Funding Information:
This study was initiated in the laboratory of Vann Bennett at the Howard Hughes Medical Institute at the Duke University Medical Center with funding provided by the Howard Hughes Medical Institute. We thank Jan Hoffman, who was integral in the isolation of the eq1 and eq2 alleles in the PCR-based deletion mutagenesis screen, the University of Minnesota C. elegans community for exchange of ideas; in particular John Yochem and Todd Starich for anatomy lessons, and Robert Herman, Mary Montgomery, Ann Rougvie, Jocelyn Shaw, Lisa Timmons, and Margaret Titus for experimental and editorial advice. We also thank Theresa Stiernagle/ C. elegans Genetics Center for providing strains. This study was supported by funding provided by the March of Dimes Basil O'Connor award (#5-FY02-245), the Minnesota Medical Foundation award (#3206-9227-02), and the University of Minnesota Grants-in-aid award (#19212).

Keywords

  • Adhesion
  • Axon
  • Caenorhabditis elegans
  • Embryo
  • L1
  • Muscle
  • Neuron
  • lad-1
  • sax-7

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