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In the zebrafish, mutation of adgrg6 also results in defects in the inner ear: otic tissue fails to down-regulate versican gene expression and morphogenesis is disrupted. We have designed a whole-animal screen that tests for rescue of both up- and down-regulated gene expression in mutant embryos, together with analysis of weak and strong alleles. From a screen of 3120 structurally diverse compounds, we have identified 68 that reduce versican b expression in the adgrg6 mutant ear, 41 of which also restore myelin basic protein gene expression in Schwann cells of mutant embryos. Nineteen compounds unable to rescue a strong adgrg6 allele provide candidates for molecules that may interact directly with the Adgrg6 receptor. Our pipeline provides a powerful approach for identifying compounds that modulate GPCR activity, with potential impact for future drug design.<\/jats:p>","DOI":"10.7554\/elife.44889","type":"journal-article","created":{"date-parts":[[2019,6,10]],"date-time":"2019-06-10T08:00:24Z","timestamp":1560153624000},"source":"Crossref","is-referenced-by-count":27,"title":["Identification of compounds that rescue otic and myelination defects in the zebrafish adgrg6 (gpr126) mutant"],"prefix":"10.7554","volume":"8","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-9336-7965","authenticated-orcid":true,"given":"Elvira","family":"Diamantopoulou","sequence":"first","affiliation":[{"name":"Bateson Centre and Department of Biomedical Science, University of Sheffield, Sheffield, United 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