Background <p>The cytoplasmic dynein complex mediates retrograde transport of various intracellular components and plays a critical role in mitosis, nuclear migration, organelle positioning, vesicle trafficking, misfolded protein clearance, and intercellular signaling.</p> Main body <p>Mutations in human cytoplasmic dynein subunits and regulators have been directly linked to various neurological diseases and are increasingly recognized as contributors to ocular degenerative diseases. However, the precise mechanisms underlying ocular morphogenesis and degeneration remain poorly understood. Various animal models, including <i>Drosophila</i>, mouse, and zebrafish, have been established to investigate the pathogenesis of cytoplasmic dynein complex-related ocular disorders. Findings from these models indicate that dynein complex-related ocular pathologies often involve endoplasmic reticulum stress, impaired Notch signaling, and disrupted Sonic Hedgehog pathways. Systematically integrating gene functional data and molecular mechanism clues derived from different models can help refine the “gene-phenotype-mechanism” correlation network and may ultimately advance precision diagnosis and targeted therapy.</p> Conclusion <p>Emerging evidence has highlighted the cytoplasmic dynein complex as a key factor in ocular development and disease. Integrating findings across model systems may enable more precise diagnosis and the development of targeted interventions for dynein-related ocular disorders.</p>