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The exact molecular function of MAB21L2 is currently unknown. We conducted a series of yeast two\u2010hybrid (Y2H) experiments to determine protein interactomes of normal human and zebrafish MAB21L2\/mab21l2 as well as human disease\u2010associated variant MAB21L2\u2010p.(Arg51Gly) using human adult retina and zebrafish embryo libraries.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>These screens identified klhl31, tnpo1, TNPO2\/tnpo2, KLC2\/klc2, and SPTBN1\/sptbn1 as co\u2010factors of MAB21L2\/mab21l2. Several factors, including hspa8 and hspa5, were found to interact with MAB21L2\u2010p.Arg51Gly but not wild\u2010type MAB21L2\/mab21l2 in Y2H screens. Further analyses via 1\u2010by\u20101 Y2H assays, co\u2010immunoprecipitation, and mass spectrometry revealed that both normal and variant MAB21L2 interact with HSPA5 and HSPA8. In situ hybridization detected co\u2010expression of <jats:italic>hspa5<\/jats:italic> and <jats:italic>hspa8<\/jats:italic> with <jats:italic>mab21l2<\/jats:italic> during eye development in zebrafish. Examination of zebrafish mutant <jats:italic>hspa8<\/jats:italic><jats:sup><jats:italic>hi138Tg<\/jats:italic><\/jats:sup> identified reduced <jats:italic>hspa8<\/jats:italic> expression associated with severe ocular developmental defects, including small eye, coloboma, and anterior segment dysgenesis. To investigate the effects of <jats:italic>hspa8<\/jats:italic> deficiency on the <jats:italic>mab21l2<\/jats:italic><jats:sup><jats:italic>Arg51_Phe52del<\/jats:italic><\/jats:sup> allele, corresponding zebrafish double mutants were generated and found to be more severely affected than single mutant lines.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>This study identifies heat shock proteins as interacting partners of MAB21L2\/mab21l2 and suggests a role for this interaction in vertebrate eye development.<\/jats:p><\/jats:sec>","DOI":"10.1002\/dvdy.560","type":"journal-article","created":{"date-parts":[[2022,12,28]],"date-time":"2022-12-28T10:08:31Z","timestamp":1672222111000},"page":"510-526","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Identification of <scp>HSPA8<\/scp> as an interacting partner of <scp>MAB21L2<\/scp> and an important factor in eye 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