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- W2566270453 abstract "Gene therapy for treating genetic hearing loss has made astonishing progress with biotechnical advance, but it still remains the difficulty to treat the disorder ultimately. A diversity of viral vectors are used in gene therapy as vehicle for delivery of therapeutic gene. Moreover, the successful outcome of the gene therapy depends on timing and delivery route as well as titer of virus. Here, we demonstrate that gene therapy by in utero is a promising tool for treating genetic hearing loss. We used adeno-associated virus serotype 1 (AAV1) to express therapeutic gene in mice. To evaluate the safety and efficiency of virus, we transferred an AAV1-GFP into the otocyst at embryonic day 12.5, and the contralateral ear was used as a control. After the gene transfer, a strong expression of transgene was observed without ototoxicity within cochlea and the hearing ability was unaffected by AAV. These results were identified by several histological interpretations and auditory brainstem responses, respectively. We also examined a successful expression of an AAV1-MsrB3-GFP within hair cells in the same way. Together, these results indicate that gene therapy by in utero is potential strategy to treat genetic hearing loss by monogenic mutations. It may apply to treat the deficiency of the MsrB3 gene which results in autosomal recessive non-syndromic hearing loss, DFNB74, in humans." @default.
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- W2566270453 date "2015-05-01" @default.
- W2566270453 modified "2023-09-25" @default.
- W2566270453 title "349. AAV-Mediated In Utero Gene Therapy To Treat Genetic Hearing Loss" @default.
- W2566270453 doi "https://doi.org/10.1016/s1525-0016(16)33958-2" @default.
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