2019
DOI: 10.1016/j.jgg.2019.01.004
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Targeted knockout of duox causes defects in zebrafish growth, thyroid development, and social interaction

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Cited by 8 publications
(10 citation statements)
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References 19 publications
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“…4B and SI Appendix, Table S6). Within the differentially expressed genes, we observed duox, which encodes a dual oxidase implicated in TH production (27,35). This gene was significantly overexpressed in new recruits from S. gigantea (adjusted P = 0.038, Log 2 FC =2.53) compared to those from H. magnifica.…”
Section: Resultsmentioning
confidence: 94%
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“…4B and SI Appendix, Table S6). Within the differentially expressed genes, we observed duox, which encodes a dual oxidase implicated in TH production (27,35). This gene was significantly overexpressed in new recruits from S. gigantea (adjusted P = 0.038, Log 2 FC =2.53) compared to those from H. magnifica.…”
Section: Resultsmentioning
confidence: 94%
“…For this, we used zebrafish Danio rerio, in which iridophores depend on TH for their maturation (22). duox requirements have been described for somatic development and melanophore numbers but not iridophore pattern (27,35). We therefore injected one-cell stage embryos of the iridophore reporter line Tg(pnp4a:palm-mcherry) wprt10Tg with highly efficient Alt-R CRISPR-Cas9 (36) targeting duox, resulting in phenotypes concordant with those for this locus (27,35) and other hypothyroid fish (21,22).…”
Section: Resultsmentioning
confidence: 99%
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“…About 105 DUOX2 variants have been reported in more than 200 unrelated CH patients (Muzza and Fugazzola, 2017). The last two years witnessed the characterization of several loss-of-function models for duox and duoxa in zebrafish including the present study (Trubiroha et al, 2018;Chopra et al, 2019;Park et al, 2019). The mutation spectrum includes single nucleotide variants as well as small deletions (frameshift as well as in-frame).…”
Section: Discussionmentioning
confidence: 80%
“…During the completion of this manuscript, two studies have reported the phenotypic consequences of duox genetic invalidation in adult zebrafish. The exon 3 of the duox gene was targeted by CRISPR/Cas9 to establish an 8 nucleotide deletion mutant fish line (Park et al, 2019). Fifty percent of adult fish carrying the biallelic duox deletion developed an externally visible goiter highlighting again the permanent goitrogenic effects of Duox inactivation.…”
Section: Discussionmentioning
confidence: 99%