Effect of Dync1h1 on Phototransduction Protein Transport and the Development and Maintenance of Photoreceptor Cells in Zebrafish.

ABSTRACT:

Purpose

Retinitis pigmentosa (RP) is a hereditary retinal disease characterized by progressive degeneration of photoreceptor cells (PRCs). Identifying potential pathogenic genes and understanding the mechanisms of PRC degeneration are essential for improving diagnosis and treatment. Cytoplasmic dynein 1, responsible for retrograde axonal transport along microtubules, plays critical roles in neuronal function. This study utilized dync1h1-deficient zebrafish to investigate its roles in PRC morphogenesis and degeneration.

Methods

Heterozygous and homozygous dync1h1-deficient zebrafish were confirmed through Sanger sequencing. Morphological changes and retinal phenotypes were assessed through histological analysis. RNA sequencing and bioinformatics were used to explore molecular

SUBMITTER: Zhou X 

PROVIDER: S-EPMC11827896 | biostudies-literature | 2025 Feb

REPOSITORIES: biostudies-literature