Preliminary evidence for enhanced auditory cortex activation and mental development after gene therapy in children with autosomal recessive deafness 9

Jiajia Zhang, Zengzhi Guo, Changjie Pan, Chunchun Hu, Xinyang Weng, Yang-wenyi Liu, Xiaoting Cheng, Jun Lv, Qi Cao, Hui Wang, Yuxin Chen, Daqi Wang, Shaowei Hu, Mengzhao Xun, Longlong Zhang, Zijing Wang, Honghai Tang, Biyun Zhu, Luo Guo, Sha Yu, Xiaoling Hu, Lin Chen, Bing Chen, Zheng-Yi Chen, Shan Sun, Xiu Xu, Huawei Li, Fei Chen and Yilai Shu ()
Additional contact information
Jiajia Zhang: Fudan University
Zengzhi Guo: Northeastern University
Changjie Pan: Southern University of Science and Technology
Chunchun Hu: Children’s Hospital of Fudan University
Xinyang Weng: Fudan University
Yang-wenyi Liu: Fudan University
Xiaoting Cheng: Fudan University
Jun Lv: Fudan University
Qi Cao: Fudan University
Hui Wang: Fudan University
Yuxin Chen: Fudan University
Daqi Wang: Fudan University
Shaowei Hu: Fudan University
Mengzhao Xun: Fudan University
Longlong Zhang: Fudan University
Zijing Wang: Fudan University
Honghai Tang: Fudan University
Biyun Zhu: Fudan University
Luo Guo: Fudan University
Sha Yu: Fudan University
Xiaoling Hu: Hong Kong Polytechnic University
Lin Chen: University of Science and Technology of China
Bing Chen: Fudan University
Zheng-Yi Chen: Harvard Medical School
Shan Sun: Fudan University
Xiu Xu: Children’s Hospital of Fudan University
Huawei Li: Fudan University
Fei Chen: Southern University of Science and Technology
Yilai Shu: Fudan University

Nature Human Behaviour, 2025, vol. 9, issue 7, 1457-1469

Abstract: Abstract Individuals with congenital deafness that have received gene therapy represent a unique group who experience hearing recovery and speech development. However, it is unclear how hearing-related cortex changes because of gene therapy. Here we study neural processing in ten patients using functional near-infrared spectroscopy and electroencephalography during a six-month follow-up period. Patients showed an enhancement of activation in the auditory cortex, particularly in parts of the Sylvian parietotemporal area while listening to music. Activation in the right anterior temporal lobe and left Sylvian parietotemporal area was also enhanced when listening to speech. The electroencephalography data showed that the power of the resting-state electroencephalography beta band at time points T2 and T3 was statistically significantly increased after gene therapy, and mismatch negativity amplitudes at T2 and T3 were statistically significantly higher than those at T0. The mental developmental level of the patients also increased after gene therapy. These preliminary findings illuminate the neural and cognitive effects of gene therapy, supporting its potential effectiveness in auditory and mental development.

Date: 2025
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DOI: 10.1038/s41562-025-02184-8

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