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Published online 7 August 2000. doi:10.1083/jcb.150.3.589
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© The Rockefeller University Press, 0021-9525/2000//589 $5.00
The Journal of Cell Biology, Volume 150, Number 3, , 2000 589-600

Original Article

Mutations in Synaptojanin Disrupt Synaptic Vesicle Recycling



Todd W. Harrisa, Erika Hartwiegb, H. Robert Horvitzb, and Erik M. Jorgensena

a Department of Biology, University of Utah, Salt Lake City, Utah 84112-0840
b Department of Biology, Massachussetts Institute of Technology, Cambridge, Massachusetts 02139
Department of Biology, University of Utah, 257 South 1400 East, Salt Lake City, UT 84112-0840.801 581-4668801 585-3517

jorgensen{at}biology.utah.edu

Synaptojanin is a polyphosphoinositide phosphatase that is found at synapses and binds to proteins implicated in endocytosis. For these reasons, it has been proposed that synaptojanin is involved in the recycling of synaptic vesicles. Here, we demonstrate that the unc-26 gene encodes the Caenorhabditis elegans ortholog of synaptojanin. unc-26 mutants exhibit defects in vesicle trafficking in several tissues, but most defects are found at synaptic termini. Specifically, we observed defects in the budding of synaptic vesicles from the plasma membrane, in the uncoating of vesicles after fission, in the recovery of vesicles from endosomes, and in the tethering of vesicles to the cytoskeleton. Thus, these results confirm studies of the mouse synaptojanin 1 mutants, which exhibit defects in the uncoating of synaptic vesicles (Cremona, O., G. Di Paolo, M.R. Wenk, A. Luthi, W.T. Kim, K. Takei, L. Daniell, Y. Nemoto, S.B. Shears, R.A. Flavell, D.A. McCormick, and P. De Camilli. 1999. Cell. 99:179–188), and further demonstrate that synaptojanin facilitates multiple steps of synaptic vesicle recycling.

Key Words: Caenorhabditis elegans • synaptic transmission • endocytosis • unc-26 • polyphosphoinositide phosphatase

© 2000 The Rockefeller University Press

Abbreviations used in this paper: Ce-synaptojanin, Caenorhabditis elegans synaptojanin; EST, expressed sequence tag; GABA, {gamma}-aminobutyric acid; GFP, green fluorescent protein; ORF, open reading frame; PI, phosphoinositide.


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