Home | Help | Feedback | Subscriptions | Archive | Search | Table of Contents

This Article Full Text Full Text (PDF, 355K) PPT slides of all figures Alert me when this article is cited Citation Map Services Email this article Similar articles in this journal Similar articles in PubMed Alert me to new content in the JCB Download to citation manager Citing Articles Citing Articles via HighWire Citing Articles via CrossRef Citing Articles via Google Scholar Google Scholar Articles by Hjalt, T. A. Articles by Murray, J. C. Search for Related Content PubMed PubMed Citation Articles by Hjalt, T. A. Articles by Murray, J. C. Social Bookmarking                            What's this?

© The Rockefeller University Press, 0021-9525/2001//545 $5.00
The Journal of Cell Biology, Volume 152, Number 3, , 2001 545-552

Pitx2 Regulates Procollagen Lysyl Hydroxylase (Plod) Gene Expression

Tord A. Hjalt^a, Brad A. Amendt^b, and Jeffrey C. Murray^a

^a Department of Pediatrics, University of Iowa, Iowa City, Iowa 52242
^b Department of Biological Science, University of Tulsa, Tulsa, Oklahoma 74104
University of Iowa, Department of Pediatrics, 140 EMRB, Iowa City, IA 52242.(319) 335-6970(319) 335-6897

jeff-murray{at}uiowa.edu

The Rieger syndrome is an autosomal dominant disease characterized by ocular, craniofacial, and umbilical defects. Patients have mutations in PITX2, a paired-bicoid homeobox gene, also involved in left/right polarity determination. In this study we have identified a family of genes for enzymes responsible for hydroxylizing lysines in collagens as one group of likely cognate targets of PITX2 transcriptional regulation. The mouse procollagen lysyl hydroxylase (Plod)-2 gene was enriched for by chromatin precipitation using a PITX2/Pitx2-specific antibody. Plod-2, as well as the human PLOD-1 promoters, contains multiple bicoid (PITX2) binding elements. We show these elements to bind PITX2 specifically in vitro. The PLOD-1 promoter induces the expression of a luciferase reporter gene in the presence of PITX2 in cotransfection experiments. The Rieger syndrome causing PITX2 mutant T68P fails to induce PLOD-1–luciferase. Mutations and rearrangements in PLOD-1 are known to be prevalent in patients with Ehlers-Danlos syndrome, kyphoscoliosis type (type VI [EDVI]). Several of the same organ systems are involved in Rieger syndrome and EDVI.

Key Words: PITX2 • PLOD • Rieger • Ehlers-Danlos • promoter

© 2001 The Rockefeller University Press

CiteULike

Complore

Connotea

Del.icio.us

Digg

Facebook

Reddit

Technorati

Twitter

This article has been cited by other articles:

• Footz, T., Idrees, F., Acharya, M., Kozlowski, K., Walter, M. A. (2009). Analysis of Mutations of the PITX2 Transcription Factor Found in Patients with Axenfeld-Rieger Syndrome. IOVS 50: 2599-2606 [Abstract] [Full Text]  
• Lamba, P., Khivansara, V., D'Alessio, A. C., Santos, M. M., Bernard, D. J. (2008). Paired-Like Homeodomain Transcription Factors 1 and 2 Regulate Follicle-Stimulating Hormone {beta}-Subunit Transcription through a Conserved cis-Element. Endocrinology 149: 3095-3108 [Abstract] [Full Text]  
• Weng, J., Luo, J., Cheng, X., Jin, C., Zhou, X., Qu, J., Tu, L., Ai, D., Li, D., Wang, J., Martin, J. F., Amendt, B. A., Liu, M. (2008). Deletion of G protein-coupled receptor 48 leads to ocular anterior segment dysgenesis (ASD) through down-regulation of Pitx2. Proc. Natl. Acad. Sci. USA 105: 6081-6086 [Abstract] [Full Text]  
• Amen, M., Espinoza, H. M., Cox, C., Liang, X., Wang, J., Link, T. M. E., Brennan, R. G., Martin, J. F., Amendt, B. A. (2008). Chromatin-associated HMG-17 is a major regulator of homeodomain transcription factor activity modulated by Wnt/{beta}-catenin signaling. Nucleic Acids Res 36: 462-476 [Abstract] [Full Text]  
• Amen, M., Liu, X., Vadlamudi, U., Elizondo, G., Diamond, E., Engelhardt, J. F., Amendt, B. A. (2007). PITX2 and {beta}-Catenin Interactions Regulate Lef-1 Isoform Expression. Mol. Cell. Biol. 27: 7560-7573 [Abstract] [Full Text]  
• Ai, D., Wang, J., Amen, M., Lu, M.-F., Amendt, B. A., Martin, J. F. (2007). Nuclear Factor 1 and T-Cell Factor/LEF Recognition Elements Regulate Pitx2 Transcription in Pituitary Development. Mol. Cell. Biol. 27: 5765-5775 [Abstract] [Full Text]  
• Gould, D. B., Marchant, J. K., Savinova, O. V., Smith, R. S., John, S. W.M. (2007). Col4a1 mutation causes endoplasmic reticulum stress and genetically modifiable ocular dysgenesis. Hum Mol Genet 16: 798-807 [Abstract] [Full Text]  
• Tamimi, Y., Skarie, J. M., Footz, T., Berry, F. B., Link, B. A., Walter, M. A. (2006). FGF19 is a target for FOXC1 regulation in ciliary body-derived cells. Hum Mol Genet 15: 3229-3240 [Abstract] [Full Text]  
• Evans, A. L., Gage, P. J. (2005). Expression of the homeobox gene Pitx2 in neural crest is required for optic stalk and ocular anterior segment development. Hum Mol Genet 14: 3347-3359 [Abstract] [Full Text]  
• Tamimi, Y., Lines, M., Coca-Prados, M., Walter, M. A. (2004). Identification of Target Genes Regulated by FOXC1 Using Nickel Agarose-Based Chromatin Enrichment. IOVS 45: 3904-3913 [Abstract] [Full Text]  
• Holmberg, J., Liu, C.-Y., Hjalt, T. A. (2004). PITX2 Gain-of-Function in Rieger Syndrome Eye Model. Am. J. Pathol. 165: 1633-1641 [Abstract] [Full Text]  
• Lines, M. A., Kozlowski, K., Kulak, S. C., Allingham, R. R., Heon, E., Ritch, R., Levin, A. V., Shields, M. B., Damji, K. F., Newlin, A., Walter, M. A. (2004). Characterization and Prevalence of PITX2 Microdeletions and Mutations in Axenfeld-Rieger Malformations. IOVS 45: 828-833 [Abstract] [Full Text]  
• Ganga, M., Espinoza, H. M., Cox, C. J., Morton, L., Hjalt, T. A., Lee, Y., Amendt, B. A. (2003). PITX2 Isoform-specific Regulation of Atrial Natriuretic Factor Expression: SYNERGISM AND REPRESSION WITH Nkx2.5. J. Biol. Chem. 278: 22437-22445 [Abstract] [Full Text]  
• Quentien, M.-H., Pitoia, F., Gunz, G., Guillet, M.-P., Enjalbert, A., Pellegrini, I. (2002). Regulation of Prolactin, GH, and Pit-1 Gene Expression in Anterior Pituitary by Pitx2: An Approach Using Pitx2 Mutants. Endocrinology 143: 2839-2851 [Abstract] [Full Text]  
• Cox, C. J., Espinoza, H. M., McWilliams, B., Chappell, K., Morton, L., Hjalt, T. A., Semina, E. V., Amendt, B. A. (2002). Differential Regulation of Gene Expression by PITX2 Isoforms. J. Biol. Chem. 277: 25001-25010 [Abstract] [Full Text]  
• Lines, M. A., Kozlowski, K., Walter, M. A. (2002). Molecular genetics of Axenfeld-Rieger malformations. Hum Mol Genet 11: 1177-1187 [Abstract] [Full Text]  
• Espinoza, H. M., Cox, C. J., Semina, E. V., Amendt, B. A. (2002). A molecular basis for differential developmental anomalies in Axenfeld-Rieger syndrome. Hum Mol Genet 11: 743-753 [Abstract] [Full Text]  
• Westmoreland, J. J., McEwen, J., Moore, B. A., Jin, Y., Condie, B. G. (2001). Conserved Function of Caenorhabditis elegans UNC-30 and Mouse Pitx2 in Controlling GABAergic Neuron Differentiation. J. Neurosci. 21: 6810-6819 [Abstract] [Full Text]  
• Wei, Q., Adelstein, R. S. (2002). Pitx2a Expression Alters Actin-Myosin Cytoskeleton and Migration of HeLa Cells through Rho GTPase Signaling. Mol. Biol. Cell 13: 683-697 [Abstract] [Full Text]  

Home | Help | Feedback | Subscriptions | Archive | Search | Table of Contents