Efficiency of gene therapy for sensorineural hearing loss in mouse model: A meta‐analysis

Abstract Objectives Sensorineural hearing loss (SNHL) is a disorder characterized by the loss or impairment of cochlear hair cells or the auditory nerve. In recent years, gene therapy has emerged as a promising approach for SNHL treatment. The objective of this study is to evaluate the impact of gen...

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Main Authors: Nevra Keskin Yilmaz, Dogukan Ozen, Rafael da Costa Monsanto, Emre Ocak, Artur Koerig Schuster, Tomotaka Shimura, Sebahattin Cureoglu
Format: Article
Language:English
Published: Wiley 2024-12-01
Series:Laryngoscope Investigative Otolaryngology
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Online Access:https://doi.org/10.1002/lio2.70048
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author Nevra Keskin Yilmaz
Dogukan Ozen
Rafael da Costa Monsanto
Emre Ocak
Artur Koerig Schuster
Tomotaka Shimura
Sebahattin Cureoglu
author_facet Nevra Keskin Yilmaz
Dogukan Ozen
Rafael da Costa Monsanto
Emre Ocak
Artur Koerig Schuster
Tomotaka Shimura
Sebahattin Cureoglu
author_sort Nevra Keskin Yilmaz
collection DOAJ
description Abstract Objectives Sensorineural hearing loss (SNHL) is a disorder characterized by the loss or impairment of cochlear hair cells or the auditory nerve. In recent years, gene therapy has emerged as a promising approach for SNHL treatment. The objective of this study is to evaluate the impact of gene therapy on the restoration or improvement of auditory function in mouse model with loss or impairment of hearing. Methods Studies with clear experimental designs, and auditory brainstem response (ABR) analysis as relevant outcome measures were included by searching PubMed, Scopus, and Web of Science databases. The PRISMA guideline was used for abstracting data and assessing data quality and validity. A quantitative synthesis was performed using a random effects model to examine the effect of gene therapy on auditory function in SNHL. Results Nine articles including 71 studies meeting the inclusion criteria were identified. These studies explored therapies targeting the TMC1, VGLUT3, USH1C, CLRN1, WHRN, and PJVK genes, with genetic material ranging from 1.8 × 1011 and 1.4 × 1014 gc/mL being delivered to the inner ear through round window membrane, cochleostomy, or posterior semicircular canal injection methods. The hearing test results showed a significant mean difference of 26.91 dB (95% CI: 22.01–31.85) in favor of the experimental group. Conclusions Although promising results have been obtained regarding the potential success of gene therapy in SNHL, further investigation is needed to explore the long‐term effects of gene therapy, treatment response rates, and the relationships between different genetic mutation types.
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spelling doaj-art-500bb52e4e3e45669f090d6f068c3c8a2025-08-20T02:39:59ZengWileyLaryngoscope Investigative Otolaryngology2378-80382024-12-0196n/an/a10.1002/lio2.70048Efficiency of gene therapy for sensorineural hearing loss in mouse model: A meta‐analysisNevra Keskin Yilmaz0Dogukan Ozen1Rafael da Costa Monsanto2Emre Ocak3Artur Koerig Schuster4Tomotaka Shimura5Sebahattin Cureoglu6Department of Internal Medicine Faculty of Veterinary Medicine, Ankara University Ankara TurkeyDepartment of Biostatistics Faculty of Veterinary Medicine, Ankara University Ankara TurkeyDepartment of Otolaryngology Head & Neck Surgery University of Minnesota Minneapolis Minnesota USADepartment of Otolaryngology Head & Neck Surgery Faculty of Medicine, Ankara University Ankara TurkeyPostgraduate Program in Medicine: Surgical Sciences, Universidade Federal do Rio Grande do Sul (UFRGS) Porto Alegre RS BrazilDepartment of Otolaryngology Head & Neck Surgery University of Minnesota Minneapolis Minnesota USADepartment of Otolaryngology Head & Neck Surgery University of Minnesota Minneapolis Minnesota USAAbstract Objectives Sensorineural hearing loss (SNHL) is a disorder characterized by the loss or impairment of cochlear hair cells or the auditory nerve. In recent years, gene therapy has emerged as a promising approach for SNHL treatment. The objective of this study is to evaluate the impact of gene therapy on the restoration or improvement of auditory function in mouse model with loss or impairment of hearing. Methods Studies with clear experimental designs, and auditory brainstem response (ABR) analysis as relevant outcome measures were included by searching PubMed, Scopus, and Web of Science databases. The PRISMA guideline was used for abstracting data and assessing data quality and validity. A quantitative synthesis was performed using a random effects model to examine the effect of gene therapy on auditory function in SNHL. Results Nine articles including 71 studies meeting the inclusion criteria were identified. These studies explored therapies targeting the TMC1, VGLUT3, USH1C, CLRN1, WHRN, and PJVK genes, with genetic material ranging from 1.8 × 1011 and 1.4 × 1014 gc/mL being delivered to the inner ear through round window membrane, cochleostomy, or posterior semicircular canal injection methods. The hearing test results showed a significant mean difference of 26.91 dB (95% CI: 22.01–31.85) in favor of the experimental group. Conclusions Although promising results have been obtained regarding the potential success of gene therapy in SNHL, further investigation is needed to explore the long‐term effects of gene therapy, treatment response rates, and the relationships between different genetic mutation types.https://doi.org/10.1002/lio2.70048gene therapymeta‐analysismicesensorineural hearing loss
spellingShingle Nevra Keskin Yilmaz
Dogukan Ozen
Rafael da Costa Monsanto
Emre Ocak
Artur Koerig Schuster
Tomotaka Shimura
Sebahattin Cureoglu
Efficiency of gene therapy for sensorineural hearing loss in mouse model: A meta‐analysis
Laryngoscope Investigative Otolaryngology
gene therapy
meta‐analysis
mice
sensorineural hearing loss
title Efficiency of gene therapy for sensorineural hearing loss in mouse model: A meta‐analysis
title_full Efficiency of gene therapy for sensorineural hearing loss in mouse model: A meta‐analysis
title_fullStr Efficiency of gene therapy for sensorineural hearing loss in mouse model: A meta‐analysis
title_full_unstemmed Efficiency of gene therapy for sensorineural hearing loss in mouse model: A meta‐analysis
title_short Efficiency of gene therapy for sensorineural hearing loss in mouse model: A meta‐analysis
title_sort efficiency of gene therapy for sensorineural hearing loss in mouse model a meta analysis
topic gene therapy
meta‐analysis
mice
sensorineural hearing loss
url https://doi.org/10.1002/lio2.70048
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