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Among the diverse pathogenic mechanisms, dysregulation of alternative splicing has emerged as a pivotal driver, particularly in RP cases caused by mutations in splicing factors or cis-regulatory elements. Alternative splicing governs transcript diversity and fine-tunes gene expression, with more than 95% of human multi-exon genes undergoing this process. Disruption of precise splicing patterns in the retina\u2014an organ with exceptionally high transcriptional complexity\u2014leads to widespread mis-splicing of photoreceptor-specific genes, triggering retinal dysfunction and cell death. This review synthesizes current understanding of alternative splicing-related mechanisms in RP, integrating molecular insights from splicing-factor mutations, retina-specific splice isoforms, and their downstream cellular consequences. We also evaluate therapeutic strategies targeting splicing dysregulation, including antisense oligonucleotides (ASOs), modified U1 snRNA, spliceosome-mediated RNA trans-splicing (SMaRT), and genome editing, emphasizing translational potential and clinical challenges. Finally, we highlight key research gaps and propose future directions for splicing-centered precision medicine in RP.<\/jats:p>","DOI":"10.3390\/biom15111624","type":"journal-article","created":{"date-parts":[[2025,11,19]],"date-time":"2025-11-19T10:02:13Z","timestamp":1763546533000},"page":"1624","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["Alternative Splicing Dysregulation in Retinitis Pigmentosa: Pathogenic Mechanisms and Therapeutic Opportunities"],"prefix":"10.3390","volume":"15","author":[{"given":"Yuxin","family":"Jiang","sequence":"first","affiliation":[{"name":"Institute of Visual Neuroscience and Stem Cell Engineering, Wuhan University of Science and Technology, Wuhan 430065, China"},{"name":"College of Life Sciences and Health, Wuhan University of Science and Technology, Wuhan 430065, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xuyu","family":"Liu","sequence":"additional","affiliation":[{"name":"Institute of Visual Neuroscience and Stem Cell Engineering, Wuhan University of Science and Technology, Wuhan 430065, China"},{"name":"College of Life Sciences and Health, Wuhan University of Science and Technology, Wuhan 430065, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jie","family":"Fu","sequence":"additional","affiliation":[{"name":"Institute of Visual Neuroscience and Stem Cell Engineering, Wuhan University of Science and Technology, Wuhan 430065, China"},{"name":"College of Life Sciences and Health, Wuhan University of Science and Technology, Wuhan 430065, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yican","family":"Wu","sequence":"additional","affiliation":[{"name":"Institute of Visual Neuroscience and Stem Cell Engineering, Wuhan University of Science and Technology, Wuhan 430065, China"},{"name":"College of Life Sciences and Health, Wuhan University of Science and Technology, Wuhan 430065, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8748-5877","authenticated-orcid":false,"given":"Shanshan","family":"Yu","sequence":"additional","affiliation":[{"name":"Institute of Visual Neuroscience and Stem Cell Engineering, Wuhan University of Science and Technology, Wuhan 430065, China"},{"name":"College of Life Sciences and Health, Wuhan University of Science and Technology, Wuhan 430065, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8122-430X","authenticated-orcid":false,"given":"Kai","family":"Yao","sequence":"additional","affiliation":[{"name":"Institute of Visual Neuroscience and Stem Cell Engineering, Wuhan University of Science and Technology, Wuhan 430065, China"},{"name":"College of Life Sciences and Health, Wuhan University of Science and Technology, Wuhan 430065, China"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2025,11,19]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"101245","DOI":"10.1016\/j.preteyeres.2024.101245","article-title":"Mechanisms of blood-retinal barrier disruption related to intraocular inflammation and malignancy","volume":"99","author":"Niederer","year":"2024","journal-title":"Prog. 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