{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,8]],"date-time":"2026-07-08T05:59:52Z","timestamp":1783490392397,"version":"3.55.0"},"reference-count":310,"publisher":"MDPI AG","issue":"18","license":[{"start":{"date-parts":[[2020,9,21]],"date-time":"2020-09-21T00:00:00Z","timestamp":1600646400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000053","name":"National Eye Institute","doi-asserted-by":"publisher","award":["024995"],"award-info":[{"award-number":["024995"]}],"id":[{"id":"10.13039\/100000053","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["IJMS"],"abstract":"<jats:p>Ion channels are membrane-spanning integral proteins expressed in multiple organs, including the eye. In the eye, ion channels are involved in various physiological processes, like signal transmission and visual processing. A wide range of mutations have been reported in the corresponding genes and their interacting subunit coding genes, which contribute significantly to an array of blindness, termed ocular channelopathies. These mutations result in either a loss- or gain-of channel functions affecting the structure, assembly, trafficking, and localization of channel proteins. A dominant-negative effect is caused in a few channels formed by the assembly of several subunits that exist as homo- or heteromeric proteins. Here, we review the role of different mutations in switching a \u201csensing\u201d ion channel to \u201cnon-sensing,\u201d leading to ocular channelopathies like Leber\u2019s congenital amaurosis 16 (LCA16), cone dystrophy, congenital stationary night blindness (CSNB), achromatopsia, bestrophinopathies, retinitis pigmentosa, etc. We also discuss the various in vitro and in vivo disease models available to investigate the impact of mutations on channel properties, to dissect the disease mechanism, and understand the pathophysiology. Innovating the potential pharmacological and therapeutic approaches and their efficient delivery to the eye for reversing a \u201cnon-sensing\u201d channel to \u201csensing\u201d would be life-changing.<\/jats:p>","DOI":"10.3390\/ijms21186925","type":"journal-article","created":{"date-parts":[[2020,9,21]],"date-time":"2020-09-21T08:18:01Z","timestamp":1600676281000},"page":"6925","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":14,"title":["Sensing through Non-Sensing Ocular Ion Channels"],"prefix":"10.3390","volume":"21","author":[{"given":"Meha","family":"Kabra","sequence":"first","affiliation":[{"name":"McPherson Eye Research Institute, University of Wisconsin-Madison, Madison, WI 53705, USA"},{"name":"Department of Pediatrics, University of Wisconsin-Madison, Madison, WI 53706, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5385-358X","authenticated-orcid":false,"given":"Bikash Ranjan","family":"Pattnaik","sequence":"additional","affiliation":[{"name":"McPherson Eye Research Institute, University of Wisconsin-Madison, Madison, WI 53705, USA"},{"name":"Department of Pediatrics, University of Wisconsin-Madison, Madison, WI 53706, USA"},{"name":"Department of Ophthalmology and Visual Sciences, University of Wisconsin-Madison, Madison, WI 53705, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2020,9,21]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"d2023","DOI":"10.2741\/krizaj","article-title":"Calcium regulation in photoreceptors","volume":"7","author":"Krizaj","year":"2002","journal-title":"Front. 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