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PMID: 11874923 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't

The Kallmann syndrome gene homolog in C. elegans is involved in epidermal morphogenesis and neurite branching.

Development (Cambridge, England) ·Vol. 129 ·No. 5 ·2002-03-00 ·Pages 1283-94

Rugarli EI, Di Schiavi E, Hilliard MA, Arbucci S, Ghezzi C, Facciolli A, Coppola G, Ballabio A, Bazzicalupo P

Abstract

Kallmann syndrome is an inherited disorder defined by the association of anosmia and hypogonadism, owing to impaired targeting and migration of olfactory axons and gonadotropin-releasing hormone secreting neurons. The gene responsible for the X-linked form of Kallmann syndrome, KAL-1, encodes a secreted protein of still elusive function. It has been proposed that KAL-1 might be involved in some aspects of olfactory axon guidance. However, the unavailability of a mouse model, and the difficulties in studying cellular and axonal migration in vertebrates have hampered an understanding of its function. We have identified the C. elegans homolog, kal-1, and document its function in vivo. We show that kal-1 is part of a mechanism by which neurons influence migration and adhesion of epidermal cells undergoing morphogenesis during ventral enclosure and male tail formation. We also show that kal-1 affects neurite outgrowth in vivo by modulating branching. Finally, we find that human KAL-1 cDNA can compensate for the loss of worm kal-1 and that overexpression of worm or human KAL-1 cDNAs in the nematode results in the same phenotypes. These data indicate functional conservation between the human and nematode proteins and establish C. elegans as a powerful animal in which to investigate KAL function in vivo. Our findings add a new player to the set of molecules, which appear to underlie both morphogenesis and axonal/neuronal navigation in vertebrates and invertebrates.

MeSH Terms
Amino Acid Sequence Animals Caenorhabditis elegans/genetics Cell Adhesion Cell Adhesion Molecules/genetics Conserved Sequence Epidermis/growth & development Extracellular Matrix Proteins Genes, Helminth Humans Kallmann Syndrome/etiology Male Molecular Sequence Data Morphogenesis Mutation Nerve Tissue Proteins Neurites/ultrastructure Sequence Homology, Amino Acid Tail/growth & development
Chemicals
ANOS1 protein, human Cell Adhesion Molecules Extracellular Matrix Proteins Nerve Tissue Proteins
Authors & Affiliations
9 authors, click to expand affiliations / ORCID
Rugarli Elena I
Telethon Institute of Genetics and Medicine (TIGEM), via P. Castellino III, 80131 Naples, Italy. rugarli@tigem.it
Di Schiavi Elia
Hilliard Massimo A
Arbucci Salvatore
Ghezzi Cristina
Facciolli Anna
Coppola Giuseppe
Ballabio Andrea
Bazzicalupo Paolo
Article Info
Journal
Development (Cambridge, England)
Abbr.
Development
ISSN
0950-1991
Published
2002-03-00
Pages
1283-94
Language
English
Region
England
NLM ID
8701744
Subset
IM
Grants
Telethon · GGP02211 · Italy
Telethon · TGM06S01 · Italy
Databases
GENBANK
AF342986
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