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Invest Ophthalmol Vis Sci
2013 Jul 30;547:5136-43. doi: 10.1167/iovs.13-12337.
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In vivo analysis of aquaporin 0 function in zebrafish: permeability regulation is required for lens transparency.
Clemens DM
,
Németh-Cahalan KL
,
Trinh L
,
Zhang T
,
Schilling TF
,
Hall JE
.
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The zebrafish lens is well suited for studies of physiology and development due to its rapid formation in the embryo and genetic accessibility. Aquaporin 0 (AQP0), a lens-specific membrane protein, is required for lens clarity. Zebrafish have two copies of AQP0 (Aqp0a and b), whereas mammals have a single, multifunctional protein. Here we demonstrate a reliable knockdown/rescue system in zebrafish and use it to provide evidence for subfunctionalization of Aqp0a and b, as well as to show that calcium-mediated regulation of Aqp0a in zebrafish lenses is necessary for transparency. Coinjection of antisense oligonucleotides and DNA rescue constructs into zebrafish embryos, followed by evaluation of the developing fish for cataracts, was used to analyze the functions of Aqp0a and b. The water permeability and regulation characteristics of each rescue protein were tested in a Xenopus oocyte swelling assay. Both copies of AQP0 are necessary for lens clarity in the zebrafish, and neither is sufficient. Water permeability is necessary but also insufficient. Phosphorylation and regulation of Aqp0a are required for its function. In the zebrafish lens, the two closely related AQP0s have acquired distinct functions that are both necessary for lens development and clarity. Regulation of AQP0 water permeability, a well-studied phenomenon in vitro, may be physiologically relevant in the living lens.
Benga,
Water channel proteins (later called aquaporins) and relatives: past, present, and future.
2009, Pubmed
Benga,
Water channel proteins (later called aquaporins) and relatives: past, present, and future.
2009,
Pubmed
Candia,
Regional distribution of the Na(+) and K(+) currents around the crystalline lens of rabbit.
2002,
Pubmed
Chandy,
Comparison of the water transporting properties of MIP and AQP1.
1997,
Pubmed
,
Xenbase
Dahm,
Development and adult morphology of the eye lens in the zebrafish.
2007,
Pubmed
Ehring,
Properties of channels reconstituted from the major intrinsic protein of lens fiber membranes.
1990,
Pubmed
Force,
Preservation of duplicate genes by complementary, degenerative mutations.
1999,
Pubmed
Froger,
Two distinct aquaporin 0s required for development and transparency of the zebrafish lens.
2010,
Pubmed
,
Xenbase
Gao,
Lens intracellular hydrostatic pressure is generated by the circulation of sodium and modulated by gap junction coupling.
2011,
Pubmed
Gold,
AKAP2 anchors PKA with aquaporin-0 to support ocular lens transparency.
2012,
Pubmed
Gonen,
Aquaporin-0 membrane junctions form upon proteolytic cleavage.
2004,
Pubmed
Gong,
Connexins in lens development and cataractogenesis.
2007,
Pubmed
Greiling,
The zebrafish lens proteome during development and aging.
2009,
Pubmed
Greiling,
Early lens development in the zebrafish: a three-dimensional time-lapse analysis.
2009,
Pubmed
Hall,
Through a glass darkly.
2012,
Pubmed
Hou,
Recapitulation of human betaB1-crystallin promoter activity in transgenic zebrafish.
2006,
Pubmed
Jensen,
Dynamic control of slow water transport by aquaporin 0: implications for hydration and junction stability in the eye lens.
2008,
Pubmed
Kalman,
AQP0-LTR of the Cat Fr mouse alters water permeability and calcium regulation of wild type AQP0.
2006,
Pubmed
,
Xenbase
Kalman,
Phosphorylation determines the calmodulin-mediated Ca2+ response and water permeability of AQP0.
2008,
Pubmed
,
Xenbase
Kumari,
Intact AQP0 performs cell-to-cell adhesion.
2009,
Pubmed
Mathias,
The lens circulation.
2007,
Pubmed
Michea,
Lens major intrinsic protein (MIP) promotes adhesion when reconstituted into large unilamellar liposomes.
1994,
Pubmed
Michea,
Biochemical evidence for adhesion-promoting role of major intrinsic protein isolated from both normal and cataractous human lenses.
1995,
Pubmed
Németh-Cahalan,
pH and calcium regulate the water permeability of aquaporin 0.
2000,
Pubmed
,
Xenbase
Németh-Cahalan,
Molecular basis of pH and Ca2+ regulation of aquaporin water permeability.
2004,
Pubmed
,
Xenbase
Posner,
A proteome map of the zebrafish (Danio rerio) lens reveals similarities between zebrafish and mammalian crystallin expression.
2008,
Pubmed
Postlethwait,
Subfunction partitioning, the teleost radiation and the annotation of the human genome.
2004,
Pubmed
Preston,
Appearance of water channels in Xenopus oocytes expressing red cell CHIP28 protein.
1992,
Pubmed
,
Xenbase
Reichow,
Allosteric mechanism of water-channel gating by Ca2+-calmodulin.
2013,
Pubmed
Reichow,
Noncanonical binding of calmodulin to aquaporin-0: implications for channel regulation.
2008,
Pubmed
Saparov,
Water and ion permeation of aquaporin-1 in planar lipid bilayers. Major differences in structural determinants and stoichiometry.
2001,
Pubmed
Schey,
Complete map and identification of the phosphorylation site of bovine lens major intrinsic protein.
1997,
Pubmed
Varadaraj,
Regulation of aquaporin water permeability in the lens.
2005,
Pubmed
,
Xenbase
Varadaraj,
Transgenic expression of AQP1 in the fiber cells of AQP0 knockout mouse: effects on lens transparency.
2010,
Pubmed
Virkki,
Cloning and functional expression of an MIP (AQP0) homolog from killifish (Fundulus heteroclitus) lens.
2001,
Pubmed
,
Xenbase
Wree,
Requirement for asparagine in the aquaporin NPA sequence signature motifs for cation exclusion.
2011,
Pubmed
,
Xenbase
Xin,
Water permeation dynamics of AqpZ: a tale of two states.
2011,
Pubmed
Zampighi,
The structural organization and protein composition of lens fiber junctions.
1989,
Pubmed
