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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...
Autores principales: | , , , , , , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Nature Publishing Group UK
2018
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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. |
format | Online Article Text |
id | pubmed-6018676 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2018 |
publisher | Nature Publishing Group UK |
record_format | MEDLINE/PubMed |
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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