Source:http://linkedlifedata.com/resource/pubmed/id/16765094
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rdf:type | |
lifeskim:mentions | |
pubmed:issue |
3
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pubmed:dateCreated |
2006-8-14
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pubmed:abstractText |
Congenital hearing deficits can be caused by a variety of genetic and acquired conditions. Complete reversal of deficits in the peripheral auditory system may require delivery of corrective genes to cochlear progenitor cells. We tested delivery of lentivirus and an array of recombinant adeno-associated viral (AAV) serotypes for efficiency and cellular specificity of transgene expression after in utero delivery to the developing mouse otocyst. Stability of expression and safety with respect to auditory function were then tested in those vectors that had the most favorable in utero cochlear transduction characteristics (AAV2/1, AAV2/8, and lentivirus). AAV2/1 was found to be the optimal vector for in utero cochlear gene transfer. It efficiently transduced progenitors giving rise to both inner and outer hair cells and supporting cells and had no adverse effect on cochlear cell differentiation. Further, it had no pathological effect on differentiated hair cells or the integrity of the auditory nerve or brain-stem nuclei as measured by auditory brain-stem response testing. AAV2/1 promises to be useful in further studies evaluating differentiation pathways of cochlear cells in health and disease and for developing gene-based therapies for congenital and acquired forms of peripheral hearing loss.
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pubmed:grant |
http://linkedlifedata.com/resource/pubmed/grant/5-P30-DK-47747-10,
http://linkedlifedata.com/resource/pubmed/grant/P30 DK047757-10,
http://linkedlifedata.com/resource/pubmed/grant/R01 DC006442 02,
http://linkedlifedata.com/resource/pubmed/grant/R01 DC006442-02,
http://linkedlifedata.com/resource/pubmed/grant/R01 DC008595-01,
http://linkedlifedata.com/resource/pubmed/grant/R01 EY010820-07,
http://linkedlifedata.com/resource/pubmed/grant/R01 EY010820-08,
http://linkedlifedata.com/resource/pubmed/grant/R01 EY010820-09,
http://linkedlifedata.com/resource/pubmed/grant/R01 EY010820-10,
http://linkedlifedata.com/resource/pubmed/grant/R01 EY010820-11,
http://linkedlifedata.com/resource/pubmed/grant/T32 DC005363
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pubmed:language |
eng
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pubmed:journal | |
pubmed:citationSubset |
IM
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pubmed:chemical | |
pubmed:status |
MEDLINE
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pubmed:month |
Sep
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pubmed:issn |
1525-0016
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pubmed:author | |
pubmed:issnType |
Print
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pubmed:volume |
14
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pubmed:owner |
NLM
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pubmed:authorsComplete |
Y
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pubmed:pagination |
328-35
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pubmed:dateRevised |
2011-9-22
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pubmed:meshHeading |
pubmed-meshheading:16765094-Animals,
pubmed-meshheading:16765094-Cell Differentiation,
pubmed-meshheading:16765094-Cochlea,
pubmed-meshheading:16765094-Dependovirus,
pubmed-meshheading:16765094-Female,
pubmed-meshheading:16765094-Fetus,
pubmed-meshheading:16765094-Gene Therapy,
pubmed-meshheading:16765094-Genetic Vectors,
pubmed-meshheading:16765094-Green Fluorescent Proteins,
pubmed-meshheading:16765094-Hair Cells, Auditory, Inner,
pubmed-meshheading:16765094-Hair Cells, Auditory, Outer,
pubmed-meshheading:16765094-Hearing Loss, Sensorineural,
pubmed-meshheading:16765094-Lentivirus,
pubmed-meshheading:16765094-Mice,
pubmed-meshheading:16765094-Microinjections,
pubmed-meshheading:16765094-Transduction, Genetic,
pubmed-meshheading:16765094-Uterus
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pubmed:year |
2006
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pubmed:articleTitle |
In vivo delivery of recombinant viruses to the fetal murine cochlea: transduction characteristics and long-term effects on auditory function.
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pubmed:affiliation |
FM Kirby Center and Scheie Eye Institute, Department of Ophthalmology, University of Pennsylvania School of Medicine, Philadelphia, PA 19104, USA.
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pubmed:publicationType |
Journal Article,
Research Support, Non-U.S. Gov't,
Research Support, N.I.H., Extramural
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