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Methylnitrosourea MNU-induced Retinal Degeneration and Regeneration in the Zebrafish: Histological and Functional Characteristics
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Role and Impact of the brsk2 Gene in Zebrafish Retinal Development and Visual Function Characterized by Behavioral,
Jingxin Deng1, Yue Li1, Meixin Hu1
1Division of Child Health Care, Children's Hospital of Fudan University, National Children's Medical Center, 399 Wanyuan Road, Shanghai 201102, China.
International Journal of Molecular Sciences
|January 28, 2026
Summary
Deleting the BRSK2 gene enhances light perception in zebrafish larvae, revealing its crucial role in retinal development and visual function. This finding offers insights into sensory processing deficits in neurodevelopmental disorders like Autism Spectrum Disorder (ASD).
Area of Science:
- Neuroscience
- Genetics
- Developmental Biology
Background:
- Vision is critical for development, and visual impairments are linked to neurodevelopmental disorders such as Autism Spectrum Disorder (ASD).
- The Brain-specific serine/threonine-protein kinase 2 (BRSK2) gene is a known high-risk gene associated with ASD.
- Understanding the genetic basis of sensory processing deficits in ASD is crucial.
Purpose of the Study:
- To investigate the role of the BRSK2 gene in zebrafish retinal photoreceptor development and visual function.
- To determine the impact of BRSK2 deletion on retinal structure and light perception.
Main Methods:
- Utilized zebrafish models with BRSK2 gene deletion (brsk2ab-/-).
- Employed behavioral assays to assess visual function.
- Conducted histological analysis for retinal structure.
- Performed transcriptomic profiling to analyze gene expression.
Main Results:
- Zebrafish larvae lacking BRSK2 (brsk2ab-/-) showed significantly enhanced light perception compared to wild-type (WT) controls.
- Retinal structure exhibited disruptions in layered organization.
- Rhodopsin expression and key opsins involved in phototransduction pathways were upregulated.
- Genes related to visual development were also markedly upregulated.
Conclusions:
- BRSK2 plays a significant role in early retinal circuit development and visual function.
- BRSK2 deletion leads to specific sensory phenotypes, including altered light perception and opsin expression.
- This research provides insights into the mechanisms of sensory abnormalities in ASD and related neurodevelopmental conditions.
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