PUBLICATION
Pannexin-2 deficiency disrupts visual pathways and leads to ocular defects in zebrafish
- Authors
- Shanbhag, R., Zoidl, G.S.O., Nakhuda, F., Sabour, S., Nauman, H., Zoidl, C., Bahl, A., Tabatabaei, N., Zoidl, G.R.
- ID
- ZDB-PUB-250324-10
- Date
- 2025
- Source
- Biochimica et biophysica acta. Molecular basis of disease : 167807167807 (Journal)
- Registered Authors
- Zoidl, Christiane, Zoidl, Georg
- Keywords
- Ocular health, Pannexins, Sensory-motor, Transcriptomics, Visual system, Zebrafish
- MeSH Terms
-
- Larva/metabolism
- Animals
- Connexins*/deficiency
- Connexins*/genetics
- Connexins*/metabolism
- Superior Colliculi/metabolism
- Zebrafish/genetics
- Zebrafish/metabolism
- Zebrafish Proteins*/deficiency
- Zebrafish Proteins*/genetics
- Zebrafish Proteins*/metabolism
- Visual Pathways*/metabolism
- Visual Pathways*/pathology
- PubMed
- 40122183 Full text @ BBA Molecular Basis of Disease
Citation
Shanbhag, R., Zoidl, G.S.O., Nakhuda, F., Sabour, S., Nauman, H., Zoidl, C., Bahl, A., Tabatabaei, N., Zoidl, G.R. (2025) Pannexin-2 deficiency disrupts visual pathways and leads to ocular defects in zebrafish. Biochimica et biophysica acta. Molecular basis of disease. :167807167807.
Abstract
Pannexin-2 (Panx2) is a unique ion channel localized to ER-mitochondria contact sites. These specialized microdomains are abundant in neurons and glia and essential for cellular signaling and metabolism. While synaptic interactions are well-studied, the role of intracellular contacts, such as those of ER-mitochondrial junctions, in neuronal function and neurodegeneration remains largely unexplored. To investigate the roles of Panx2 in neuronal communication, we examined its expression pattern in the zebrafish brain and used TALEN technology to generate homozygous Panx2 knockout (Panx2Δ11) zebrafish. Our results demonstrate that panx2 mRNA is present in several brain regions, notably in visual centers such as the optic tectum and the thalamus. In 6 days post fertilization TL (Panx2+/+) larvae, Panx2 expression was observed in the retina and the arborization fields of the optic tract. Transcriptome profiling of Panx2Δ11 larvae by RNA-seq analysis revealed down-regulation of genes involved in visual perception and lens development. Behavioral tests showed that loss of Panx2 leads to an altered ability to interpret visual information, such as changes in ambient illuminations, and respond with the characteristic motor action. Additionally, the knockout larvae displayed significantly impaired optomotor response. Lastly, when we tested the retinal structure of adult zebrafish eyes using optical coherence tomography, Panx2Δ11 fish revealed a longer mean axial length and a negative shift in retinal refractive error (RRE) values. Our findings highlight a distinct, novel function of Panx2 in sensory perception and ocular health, beyond its recognized roles in neurodevelopment and cancer.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping