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How ATP7B dysfunction and the subsequent Cu overload induce retinal damage, however, are unknown. Here, we show that atp7b\u2212\/\u2212 homozygous zebrafish larvae are insensitive to light stimulation, with a reduction in retinal cells but normal like morphological phenotypes. Additionally, a series of differentially expressed genes are unveiled in atp7b\u2212\/\u2212 mutated larvae, which enrich in photo-transduction, structural constituent of eye lens, sensory perception of light stimulus, oxidative phosphorylation, and ATPase activity. Moreover, we show the Cu accumulation in retinal cells in atp7b\u2212\/\u2212 mutated larvae, which results in endoplasmic reticulum (ER) stress and retinal cell apoptosis and subsequent retinal defects. The integral data in this study demonstrate that atp7b mutation leads to Cu accumulation in zebrafish retinal cells and the consequence ER stress and retinal cell death. These data may give some possible hints to explain retinal disease occurred in the Cu dysregulation syndromes Wilson\u2019s disease with ATP7B mutation.<\/jats:p>","DOI":"10.1093\/mtomcs\/mfad024","type":"journal-article","created":{"date-parts":[[2023,4,18]],"date-time":"2023-04-18T13:51:11Z","timestamp":1681825871000},"source":"Crossref","is-referenced-by-count":6,"title":["<i>Atp7b<\/i> deficiency induces zebrafish eye developmental defects"],"prefix":"10.1093","volume":"15","author":[{"given":"You","family":"Wu","sequence":"first","affiliation":[{"name":"College of Fisheries, Key Laboratory of Freshwater Animal Breeding, Ministry of Agriculture, Huazhong Agricultural University , Wuhan 430070 , China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wenye","family":"Liu","sequence":"additional","affiliation":[{"name":"College of Fisheries, Key Laboratory of Freshwater Animal Breeding, Ministry of Agriculture, Huazhong Agricultural University , Wuhan 430070 , China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lingya","family":"Li","sequence":"additional","affiliation":[{"name":"College of Fisheries, Key Laboratory of Freshwater Animal Breeding, Ministry of Agriculture, Huazhong Agricultural University , Wuhan 430070 , China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zhipeng","family":"Tai","sequence":"additional","affiliation":[{"name":"College of Fisheries, Key Laboratory of Freshwater Animal Breeding, Ministry of Agriculture, Huazhong Agricultural University , Wuhan 430070 , China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"BaoXiang","family":"Gao","sequence":"additional","affiliation":[{"name":"Key Laboratory of Analytical Science and Technology of Hebei Province, College of Chemistry and Environmental Science, Hebei University , Baoding, Hebei 071002 , China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jing-Xia","family":"Liu","sequence":"additional","affiliation":[{"name":"College of Fisheries, Key Laboratory of Freshwater Animal Breeding, Ministry of Agriculture, Huazhong Agricultural University , Wuhan 430070 , China"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2023,4,18]]},"reference":[{"issue":"5","key":"2024012010405520600_bib1","doi-asserted-by":"crossref","first-page":"1579S\u20131580S","DOI":"10.1093\/jn\/133.5.1579S","article-title":"Integrating Trace Element Metabolism from the Cell to the Whole Organism","volume":"133","author":"Thiele","year":"2003","journal-title":"J. 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