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Functional Specialization of Sensory Cilia by an RFX Transcription Factor Isoform

Wang, Juan and Schwartz, Hillel T. and Barr, Maureen M. (2010) Functional Specialization of Sensory Cilia by an RFX Transcription Factor Isoform. Genetics, 186 (4). pp. 1295-1307. ISSN 0016-6731. PMCID PMC2998312.

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In animals, RFX transcription factors govern ciliogenesis by binding to an X-box motif in the promoters of ciliogenic genes. In Caenorhabditis elegans, the sole RFX transcription factor (TF) daf-19 null mutant lacks all sensory cilia, fails to express many ciliogenic genes, and is defective in many sensory behaviors, including male mating. The daf-19c isoform is expressed in all ciliated sensory neurons and is necessary and sufficient for activating X-box containing ciliogenesis genes. Here, we describe the daf-19(n4132) mutant that is defective in expression of the sensory polycystic kidney disease (PKD) gene battery and male mating behavior, without affecting expression of ciliogenic genes or ciliogenesis. daf-19(n4132) disrupts expression of a new isoform, daf-19m (for function in male mating). daf-19m is expressed in male-specific PKD and core IL2 neurons via internal promoters and remote enhancer elements located in introns of the daf-19 genomic locus. daf-19m genetically programs the sensory functions of a subset of ciliated neurons, independent of daf-19c. In the male-specific HOB neuron, DAF-19M acts downstream of the zinc finger TF EGL-46, indicating that a TF cascade controls the PKD gene battery in this cell-type specific context. We conclude that the RFX TF DAF-19 regulates ciliogenesis via X-box containing ciliogenic genes and controls ciliary specialization by regulating non-X-box containing sensory genes. This study reveals a more extensive role for RFX TFs in generating fully functional cilia.

Item Type:Article
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URLURL TypeDescription CentralArticle Information
Schwartz, Hillel T.0000-0002-3448-8652
Barr, Maureen M.0000-0003-4483-2952
Additional Information:© 2010 by the Genetics Society of America. Manuscript received September 2, 2010. Accepted for publication October 4, 2010. We thank H. R. Horvitz, in whose lab the n4132 mutant was isolated; P. Swoboda and G. Senti for sharing unpublished data and stimulating discussions; P. Anderson, J. Kimble, Q. Mitrovich, and P. W. Sternberg for critical intellectual input in the early stages of this work; Barr and Swoboda lab members for constructive criticism of the manuscript; A. Hart, M. Leroux, D. Riddle, P. W. Sternberg, and the Caenorhabditis Genetics Center, which is funded by the National Institutes of Health (NIH) National Center for Research Resources (NCRR) for strains. We also thank the two anonymous reviewers for constructive criticism and valuable experimental suggestions. This research was supported by grants from the Polycystic Kidney Disease Foundation (to J.W.) and NIH/National Institute of Diabetes and Digestive and Kidney Diseases (to M.M.B.).
Funding AgencyGrant Number
Polycystic Kidney Disease FoundationUNSPECIFIED
Issue or Number:4
PubMed Central ID:PMC2998312
Record Number:CaltechAUTHORS:20200608-115354059
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Official Citation:Functional Specialization of Sensory Cilia by an RFX Transcription Factor Isoform Juan Wang, Hillel T. Schwartz and Maureen M. Barr GENETICS December 1, 2010 vol. 186 no. 4 1295-1307;
Usage Policy:No commercial reproduction, distribution, display or performance rights in this work are provided.
ID Code:103763
Deposited By: George Porter
Deposited On:08 Jun 2020 22:57
Last Modified:08 Jun 2020 22:57

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