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In utero nanoparticle delivery for site-specific genome editing

Genetic diseases can be diagnosed early during pregnancy, but many monogenic disorders continue to cause considerable neonatal and pediatric morbidity and mortality. Early intervention through intrauterine gene editing, however, could correct the genetic defect, potentially allowing for normal organ...

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Autores principales: Ricciardi, Adele S., Bahal, Raman, Farrelly, James S., Quijano, Elias, Bianchi, Anthony H., Luks, Valerie L., Putman, Rachael, López-Giráldez, Francesc, Coşkun, Süleyman, Song, Eric, Liu, Yanfeng, Hsieh, Wei-Che, Ly, Danith H., Stitelman, David H., Glazer, Peter M., Saltzman, W. Mark
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Nature Publishing Group UK 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6018676/
https://www.ncbi.nlm.nih.gov/pubmed/29946143
http://dx.doi.org/10.1038/s41467-018-04894-2
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author Ricciardi, Adele S.
Bahal, Raman
Farrelly, James S.
Quijano, Elias
Bianchi, Anthony H.
Luks, Valerie L.
Putman, Rachael
López-Giráldez, Francesc
Coşkun, Süleyman
Song, Eric
Liu, Yanfeng
Hsieh, Wei-Che
Ly, Danith H.
Stitelman, David H.
Glazer, Peter M.
Saltzman, W. Mark
author_facet Ricciardi, Adele S.
Bahal, Raman
Farrelly, James S.
Quijano, Elias
Bianchi, Anthony H.
Luks, Valerie L.
Putman, Rachael
López-Giráldez, Francesc
Coşkun, Süleyman
Song, Eric
Liu, Yanfeng
Hsieh, Wei-Che
Ly, Danith H.
Stitelman, David H.
Glazer, Peter M.
Saltzman, W. Mark
author_sort Ricciardi, Adele S.
collection PubMed
description Genetic diseases can be diagnosed early during pregnancy, but many monogenic disorders continue to cause considerable neonatal and pediatric morbidity and mortality. Early intervention through intrauterine gene editing, however, could correct the genetic defect, potentially allowing for normal organ development, functional disease improvement, or cure. Here we demonstrate safe intravenous and intra-amniotic administration of polymeric nanoparticles to fetal mouse tissues at selected gestational ages with no effect on survival or postnatal growth. In utero introduction of nanoparticles containing peptide nucleic acids (PNAs) and donor DNAs corrects a disease-causing mutation in the β-globin gene in a mouse model of human β-thalassemia, yielding sustained postnatal elevation of blood hemoglobin levels into the normal range, reduced reticulocyte counts, reversal of splenomegaly, and improved survival, with no detected off-target mutations in partially homologous loci. This work may provide the basis for a safe and versatile method of fetal gene editing for human monogenic disorders.
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spelling pubmed-60186762018-06-27 In utero nanoparticle delivery for site-specific genome editing Ricciardi, Adele S. Bahal, Raman Farrelly, James S. Quijano, Elias Bianchi, Anthony H. Luks, Valerie L. Putman, Rachael López-Giráldez, Francesc Coşkun, Süleyman Song, Eric Liu, Yanfeng Hsieh, Wei-Che Ly, Danith H. Stitelman, David H. Glazer, Peter M. Saltzman, W. Mark Nat Commun Article Genetic diseases can be diagnosed early during pregnancy, but many monogenic disorders continue to cause considerable neonatal and pediatric morbidity and mortality. Early intervention through intrauterine gene editing, however, could correct the genetic defect, potentially allowing for normal organ development, functional disease improvement, or cure. Here we demonstrate safe intravenous and intra-amniotic administration of polymeric nanoparticles to fetal mouse tissues at selected gestational ages with no effect on survival or postnatal growth. In utero introduction of nanoparticles containing peptide nucleic acids (PNAs) and donor DNAs corrects a disease-causing mutation in the β-globin gene in a mouse model of human β-thalassemia, yielding sustained postnatal elevation of blood hemoglobin levels into the normal range, reduced reticulocyte counts, reversal of splenomegaly, and improved survival, with no detected off-target mutations in partially homologous loci. This work may provide the basis for a safe and versatile method of fetal gene editing for human monogenic disorders. Nature Publishing Group UK 2018-06-26 /pmc/articles/PMC6018676/ /pubmed/29946143 http://dx.doi.org/10.1038/s41467-018-04894-2 Text en © The Author(s) 2018 Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons license and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/.
spellingShingle Article
Ricciardi, Adele S.
Bahal, Raman
Farrelly, James S.
Quijano, Elias
Bianchi, Anthony H.
Luks, Valerie L.
Putman, Rachael
López-Giráldez, Francesc
Coşkun, Süleyman
Song, Eric
Liu, Yanfeng
Hsieh, Wei-Che
Ly, Danith H.
Stitelman, David H.
Glazer, Peter M.
Saltzman, W. Mark
In utero nanoparticle delivery for site-specific genome editing
title In utero nanoparticle delivery for site-specific genome editing
title_full In utero nanoparticle delivery for site-specific genome editing
title_fullStr In utero nanoparticle delivery for site-specific genome editing
title_full_unstemmed In utero nanoparticle delivery for site-specific genome editing
title_short In utero nanoparticle delivery for site-specific genome editing
title_sort in utero nanoparticle delivery for site-specific genome editing
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6018676/
https://www.ncbi.nlm.nih.gov/pubmed/29946143
http://dx.doi.org/10.1038/s41467-018-04894-2
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