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Differential Developmental Deficits in Retinal Function in the Absence of either Protein Tyrosine Sulfotransferase-1 or -2
To investigate the role(s) of protein-tyrosine sulfation in the retina and to determine the differential role(s) of tyrosylprotein sulfotransferases (TPST) 1 and 2 in vision, retinal function and structure were examined in mice lacking TPST-1 or TPST-2. Despite the normal histologic retinal appearan...
Autores principales: | , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Public Library of Science
2012
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3382163/ https://www.ncbi.nlm.nih.gov/pubmed/22745813 http://dx.doi.org/10.1371/journal.pone.0039702 |
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author | Sherry, David M. Kanan, Yogita Hamilton, Robert Hoffhines, Adam Arbogast, Kelsey L. Fliesler, Steven J. Naash, Muna I. Moore, Kevin L. Al-Ubaidi, Muayyad R. |
author_facet | Sherry, David M. Kanan, Yogita Hamilton, Robert Hoffhines, Adam Arbogast, Kelsey L. Fliesler, Steven J. Naash, Muna I. Moore, Kevin L. Al-Ubaidi, Muayyad R. |
author_sort | Sherry, David M. |
collection | PubMed |
description | To investigate the role(s) of protein-tyrosine sulfation in the retina and to determine the differential role(s) of tyrosylprotein sulfotransferases (TPST) 1 and 2 in vision, retinal function and structure were examined in mice lacking TPST-1 or TPST-2. Despite the normal histologic retinal appearance in both Tpst1(−/−) and Tpst2(−/−) mice, retinal function was compromised during early development. However, Tpst1(−/−) retinas became electrophysiologically normal by postnatal day 90 while Tpst2(−/−) mice did not functionally normalize with age. Ultrastructurally, the absence of TPST-1 or TPST-2 caused minor reductions in neuronal plexus. These results demonstrate the functional importance of protein-tyrosine sulfation for proper development of the retina and suggest that the different phenotypes resulting from elimination of either TPST-1 or -2 may reflect differential expression patterns or levels of the enzymes. Furthermore, single knock-out mice of either TPST-1 or -2 did not phenocopy mice with double-knockout of both TPSTs, suggesting that the functions of the TPSTs are at least partially redundant, which points to the functional importance of these enzymes in the retina. |
format | Online Article Text |
id | pubmed-3382163 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2012 |
publisher | Public Library of Science |
record_format | MEDLINE/PubMed |
spelling | pubmed-33821632012-06-28 Differential Developmental Deficits in Retinal Function in the Absence of either Protein Tyrosine Sulfotransferase-1 or -2 Sherry, David M. Kanan, Yogita Hamilton, Robert Hoffhines, Adam Arbogast, Kelsey L. Fliesler, Steven J. Naash, Muna I. Moore, Kevin L. Al-Ubaidi, Muayyad R. PLoS One Research Article To investigate the role(s) of protein-tyrosine sulfation in the retina and to determine the differential role(s) of tyrosylprotein sulfotransferases (TPST) 1 and 2 in vision, retinal function and structure were examined in mice lacking TPST-1 or TPST-2. Despite the normal histologic retinal appearance in both Tpst1(−/−) and Tpst2(−/−) mice, retinal function was compromised during early development. However, Tpst1(−/−) retinas became electrophysiologically normal by postnatal day 90 while Tpst2(−/−) mice did not functionally normalize with age. Ultrastructurally, the absence of TPST-1 or TPST-2 caused minor reductions in neuronal plexus. These results demonstrate the functional importance of protein-tyrosine sulfation for proper development of the retina and suggest that the different phenotypes resulting from elimination of either TPST-1 or -2 may reflect differential expression patterns or levels of the enzymes. Furthermore, single knock-out mice of either TPST-1 or -2 did not phenocopy mice with double-knockout of both TPSTs, suggesting that the functions of the TPSTs are at least partially redundant, which points to the functional importance of these enzymes in the retina. Public Library of Science 2012-06-22 /pmc/articles/PMC3382163/ /pubmed/22745813 http://dx.doi.org/10.1371/journal.pone.0039702 Text en Sherry et al. http://creativecommons.org/licenses/by/4.0/ This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are properly credited. |
spellingShingle | Research Article Sherry, David M. Kanan, Yogita Hamilton, Robert Hoffhines, Adam Arbogast, Kelsey L. Fliesler, Steven J. Naash, Muna I. Moore, Kevin L. Al-Ubaidi, Muayyad R. Differential Developmental Deficits in Retinal Function in the Absence of either Protein Tyrosine Sulfotransferase-1 or -2 |
title | Differential Developmental Deficits in Retinal Function in the Absence of either Protein Tyrosine Sulfotransferase-1 or -2 |
title_full | Differential Developmental Deficits in Retinal Function in the Absence of either Protein Tyrosine Sulfotransferase-1 or -2 |
title_fullStr | Differential Developmental Deficits in Retinal Function in the Absence of either Protein Tyrosine Sulfotransferase-1 or -2 |
title_full_unstemmed | Differential Developmental Deficits in Retinal Function in the Absence of either Protein Tyrosine Sulfotransferase-1 or -2 |
title_short | Differential Developmental Deficits in Retinal Function in the Absence of either Protein Tyrosine Sulfotransferase-1 or -2 |
title_sort | differential developmental deficits in retinal function in the absence of either protein tyrosine sulfotransferase-1 or -2 |
topic | Research Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3382163/ https://www.ncbi.nlm.nih.gov/pubmed/22745813 http://dx.doi.org/10.1371/journal.pone.0039702 |
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