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PAX6 MiniPromoters drive restricted expression from rAAV in the adult mouse retina

Current gene therapies predominantly use small, strong, and readily available ubiquitous promoters. However, as the field matures, the availability of small, cell-specific promoters would be greatly beneficial. Here we design seven small promoters from the human paired box 6 (PAX6) gene and test the...

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Autores principales: Hickmott, Jack W, Chen, Chih-yu, Arenillas, David J, Korecki, Andrea J, Lam, Siu Ling, Molday, Laurie L, Bonaguro, Russell J, Zhou, Michelle, Chou, Alice Y, Mathelier, Anthony, Boye, Sanford L, Hauswirth, William W, Molday, Robert S, Wasserman, Wyeth W, Simpson, Elizabeth M
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Nature Publishing Group 2016
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4980111/
https://www.ncbi.nlm.nih.gov/pubmed/27556059
http://dx.doi.org/10.1038/mtm.2016.51
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author Hickmott, Jack W
Chen, Chih-yu
Arenillas, David J
Korecki, Andrea J
Lam, Siu Ling
Molday, Laurie L
Bonaguro, Russell J
Zhou, Michelle
Chou, Alice Y
Mathelier, Anthony
Boye, Sanford L
Hauswirth, William W
Molday, Robert S
Wasserman, Wyeth W
Simpson, Elizabeth M
author_facet Hickmott, Jack W
Chen, Chih-yu
Arenillas, David J
Korecki, Andrea J
Lam, Siu Ling
Molday, Laurie L
Bonaguro, Russell J
Zhou, Michelle
Chou, Alice Y
Mathelier, Anthony
Boye, Sanford L
Hauswirth, William W
Molday, Robert S
Wasserman, Wyeth W
Simpson, Elizabeth M
author_sort Hickmott, Jack W
collection PubMed
description Current gene therapies predominantly use small, strong, and readily available ubiquitous promoters. However, as the field matures, the availability of small, cell-specific promoters would be greatly beneficial. Here we design seven small promoters from the human paired box 6 (PAX6) gene and test them in the adult mouse retina using recombinant adeno-associated virus. We chose the retina due to previous successes in gene therapy for blindness, and the PAX6 gene since it is: well studied; known to be driven by discrete regulatory regions; expressed in therapeutically interesting retinal cell types; and mutated in the vision-loss disorder aniridia, which is in need of improved therapy. At the PAX6 locus, 31 regulatory regions were bioinformatically predicted, and nine regulatory regions were constructed into seven MiniPromoters. Driving Emerald GFP, these MiniPromoters were packaged into recombinant adeno-associated virus, and injected intravitreally into postnatal day 14 mice. Four MiniPromoters drove consistent retinal expression in the adult mouse, driving expression in combinations of cell-types that endogenously express Pax6: ganglion, amacrine, horizontal, and Müller glia. Two PAX6-MiniPromoters drive expression in three of the four cell types that express PAX6 in the adult mouse retina. Combined, they capture all four cell types, making them potential tools for research, and PAX6-gene therapy for aniridia.
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spelling pubmed-49801112016-08-23 PAX6 MiniPromoters drive restricted expression from rAAV in the adult mouse retina Hickmott, Jack W Chen, Chih-yu Arenillas, David J Korecki, Andrea J Lam, Siu Ling Molday, Laurie L Bonaguro, Russell J Zhou, Michelle Chou, Alice Y Mathelier, Anthony Boye, Sanford L Hauswirth, William W Molday, Robert S Wasserman, Wyeth W Simpson, Elizabeth M Mol Ther Methods Clin Dev Article Current gene therapies predominantly use small, strong, and readily available ubiquitous promoters. However, as the field matures, the availability of small, cell-specific promoters would be greatly beneficial. Here we design seven small promoters from the human paired box 6 (PAX6) gene and test them in the adult mouse retina using recombinant adeno-associated virus. We chose the retina due to previous successes in gene therapy for blindness, and the PAX6 gene since it is: well studied; known to be driven by discrete regulatory regions; expressed in therapeutically interesting retinal cell types; and mutated in the vision-loss disorder aniridia, which is in need of improved therapy. At the PAX6 locus, 31 regulatory regions were bioinformatically predicted, and nine regulatory regions were constructed into seven MiniPromoters. Driving Emerald GFP, these MiniPromoters were packaged into recombinant adeno-associated virus, and injected intravitreally into postnatal day 14 mice. Four MiniPromoters drove consistent retinal expression in the adult mouse, driving expression in combinations of cell-types that endogenously express Pax6: ganglion, amacrine, horizontal, and Müller glia. Two PAX6-MiniPromoters drive expression in three of the four cell types that express PAX6 in the adult mouse retina. Combined, they capture all four cell types, making them potential tools for research, and PAX6-gene therapy for aniridia. Nature Publishing Group 2016-08-10 /pmc/articles/PMC4980111/ /pubmed/27556059 http://dx.doi.org/10.1038/mtm.2016.51 Text en Copyright © 2016 Official journal of the American Society of Gene & Cell Therapy http://creativecommons.org/licenses/by-nc-sa/4.0/ This work is licensed under a Creative Commons Attribution-NonCommercial-ShareAlike 4.0 International License. The images or other third party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in the credit line; if the material is not included under the Creative Commons license, users will need to obtain permission from the license holder to reproduce the material. To view a copy of this license, visit http://creativecommons.org/licenses/by-nc-sa/4.0/
spellingShingle Article
Hickmott, Jack W
Chen, Chih-yu
Arenillas, David J
Korecki, Andrea J
Lam, Siu Ling
Molday, Laurie L
Bonaguro, Russell J
Zhou, Michelle
Chou, Alice Y
Mathelier, Anthony
Boye, Sanford L
Hauswirth, William W
Molday, Robert S
Wasserman, Wyeth W
Simpson, Elizabeth M
PAX6 MiniPromoters drive restricted expression from rAAV in the adult mouse retina
title PAX6 MiniPromoters drive restricted expression from rAAV in the adult mouse retina
title_full PAX6 MiniPromoters drive restricted expression from rAAV in the adult mouse retina
title_fullStr PAX6 MiniPromoters drive restricted expression from rAAV in the adult mouse retina
title_full_unstemmed PAX6 MiniPromoters drive restricted expression from rAAV in the adult mouse retina
title_short PAX6 MiniPromoters drive restricted expression from rAAV in the adult mouse retina
title_sort pax6 minipromoters drive restricted expression from raav in the adult mouse retina
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4980111/
https://www.ncbi.nlm.nih.gov/pubmed/27556059
http://dx.doi.org/10.1038/mtm.2016.51
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