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Originally published as MBC in Press, 10.1091/mbc.E08-04-0416 on October 8, 2008

Vol. 19, Issue 12, 5517-5528, December 2008

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Distinct Isoforms of the RFX Transcription Factor DAF-19 Regulate Ciliogenesis and Maintenance of Synaptic Activity

Gabriele Senti, and Peter Swoboda

Department of Biosciences and Nutrition, Karolinska Institute, S-14157 Huddinge, Sweden; and School of Life Sciences, Södertörn University College, S-14189 Huddinge, Sweden

Submitted April 23, 2008; Revised August 15, 2008; Accepted September 30, 2008
Monitoring Editor: Marcos Gonzalez-Gaitan

Neurons form elaborate subcellular structures such as dendrites, axons, cilia, and synapses to receive signals from their environment and to transmit them to the respective target cells. In the worm Caenorhabditis elegans, lack of the RFX transcription factor DAF-19 leads to the absence of cilia normally found on 60 sensory neurons. We now describe and functionally characterize three different isoforms of DAF-19. The short isoform DAF-19C is specifically expressed in ciliated sensory neurons and sufficient to rescue all cilia-related phenotypes of daf-19 mutants. In contrast, the long isoforms DAF-19A/B function in basically all nonciliated neurons. We discovered behavioral and cellular phenotypes in daf-19 mutants that depend on the isoforms daf-19a/b. These novel synaptic maintenance phenotypes are reminiscent of synaptic decline seen in many human neurodegenerative disorders. The C. elegans daf-19 mutant worms can thus serve as a molecular model for the mechanisms of functional neuronal decline.


This was published online ahead of print in MBC in Press (http://www.molbiolcell.org/cgi/doi/10.1091/mbc.E08-04-0416) on October 8, 2008.

Address correspondence to: Peter Swoboda (peter.swoboda{at}ki.se).




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