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Am. J. Respir. Cell Mol. Biol., Volume 23, Number 1, July, 2000 45-51

Ciliogenesis and Left-Right Axis Defects in Forkhead Factor HFH-4-Null Mice

Steven L. Brody, Xiu Hua Yan, Mary K. Wuerffel, Sheng-Kwei Song, and Steven D. Shapiro

Departments of Medicine, Chemistry, and Cell Biology and Physiology, Washington University School of Medicine, St. Louis, Missouri

Cilia have been classified as sensory or motile types on the basis of functional and structural characteristics; however, factors important for regulation of assembly of different cilia types are not well understood. Hepatocyte nuclear factor-3/forkhead homologue 4 (HFH-4) is a winged helix/forkhead transcription factor expressed in ciliated cells of the respiratory tract, oviduct, and ependyma in late development through adulthood. Targeted deletion of the Hfh4 gene resulted in defective ciliogenesis in airway epithelial cells and randomized left-right asymmetry so that half the mice had situs inversus. In HFH-4-null mice, classic motile type cilia with a 9 + 2 microtubule ultrastructure were absent in epithelial cells, including those in the airways. In other organs, sensory cilia with a 9 + 0 microtubule pattern, such as those on olfactory neuroepithelial cells, were present. Ultrastructural analysis of mutant cells with absent 9 + 2 cilia demonstrated that defective ciliogenesis was due to abnormal centriole migration and/or apical membrane docking, suggesting that HFH-4 functions to direct basal body positioning or anchoring. Evaluation of wild-type embryos at gestational days 7.0 to 7.5 revealed Hfh4 expression in embryonic node cells that have monocilium, consistent with a function for this factor at the node in early determination of left- right axis. Analysis of the node of HFH-4 mutant embryos revealed that, in contrast to absent airway cilia, node cilia were present. These observations indicate that there are independent regulatory pathways for node ciliogenesis compared with 9 + 2 type ciliogenesis in airways, and support a central role for HFH-4 in ciliogenesis and left-right axis formation.




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[Abstract] [Full Text] [PDF]

Home page
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Home page
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[Abstract] [Full Text] [PDF]

Home page
 J. Cell Sci.Home page
B. N. Gomperts, X. Gong-Cooper, and B. P. Hackett
Foxj1 regulates basal body anchoring to the cytoskeleton of ciliated pulmonary epithelial cells
J. Cell Sci., March 15, 2004; 117(8): 1329 - 1337.
[Abstract] [Full Text] [PDF]

Home page
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[Abstract] [Full Text] [PDF]

Home page
 J. Cell Sci.Home page
T. Huang, Y. You, M. S. Spoor, E. J. Richer, Vrinda. V. Kudva, R. C. Paige, M. P. Seiler, J. M. Liebler, J. Zabner, C. G. Plopper, et al.
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Home page
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Home page
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[Abstract] [Full Text] [PDF]

Home page
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Hum. Mol. Genet., April 2, 2003; 12(90001): R27 - 35.
[Abstract] [Full Text] [PDF]

Home page
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Y. You, E. J. Richer, T. Huang, and S. L. Brody
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[Abstract] [Full Text] [PDF]

Home page
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Am. J. Respir. Cell Mol. Biol., August 1, 2002; 27(2): 133 - 140.
[Abstract] [Full Text] [PDF]

Home page
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Effects of Paramyxoviral Infection on Airway Epithelial Cell Foxj1 Expression, Ciliogenesis, and Mucociliary Function
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[Abstract] [Full Text] [PDF]

Home page
 Am. J. Physiol. Lung Cell. Mol. Physiol.Home page
R. H. Costa, V. V. Kalinichenko, and L. Lim
Transcription factors in mouse lung development and function
Am J Physiol Lung Cell Mol Physiol, May 1, 2001; 280(5): L823 - L838.
[Abstract] [Full Text] [PDF]

Home page
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