{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,14]],"date-time":"2026-10-14T12:57:43Z","timestamp":1791982663061,"version":"4.4.0"},"reference-count":88,"publisher":"The Company of Biologists","license":[{"start":{"date-parts":[[2021,5,18]],"date-time":"2021-05-18T00:00:00Z","timestamp":1621296000000},"content-version":"am","delay-in-days":868,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/501100000265","name":"Medical Research Council","doi-asserted-by":"publisher","award":["MR\/L007525\/1"],"award-info":[{"award-number":["MR\/L007525\/1"]}],"id":[{"id":"10.13039\/501100000265","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"abstract":"<jats:p>Thyroid dyshormonogenesis is a leading cause of congenital hypothyroidism, a highly prevalent but treatable condition. Thyroid hormone synthesis is dependent on the formation of reactive oxygen species (ROS). In humans, the primary sources for ROS production during thyroid hormone synthesis are the NADPH oxidases DUOX1 and DUOX2. Indeed, mutations in DUOX1 and DUOX2 have been linked with congenital hypothyroidism. Unlike humans, zebrafish has a single orthologue for DUOX1 and DUOX2. In this study, we investigated the phenotypes associated with two nonsense mutant alleles, sa9892 and sa13017, of the single duox gene in zebrafish. Both alleles gave rise to readily observable phenotypes reminiscent of congenital hypothyroidism, from the larval stages through to adulthood. By using various methods to examine external and internal phenotypes, we discovered a strong correlation between TH synthesis and duox function, beginning from an early larval stage, when T4 levels are already noticeably absent in the mutants. Loss of T4 production resulted in growth retardation, pigmentation defects, ragged fins, thyroid hyperplasia \/ external goiter, and infertility. Remarkably, all of these defects associated with chronic congenital hypothyroidism could be rescued with T4 treatment, even when initiated when the fish had already reached adulthood. Our work suggests that these zebrafish duox mutants may provide a powerful model to understand the aetiology of untreated and treated congenital hypothyroidism even in advance stages of development.<\/jats:p>","DOI":"10.1242\/bio.037655","type":"journal-article","created":{"date-parts":[[2019,1,30]],"date-time":"2019-01-30T10:54:22Z","timestamp":1548845662000},"source":"Crossref","is-referenced-by-count":25,"title":["Zebrafish\n                    <i>duox<\/i>\n                    mutations provide a model for human congenital hypothyroidism"],"prefix":"10.1242","author":[{"given":"Kunal","family":"Chopra","sequence":"first","affiliation":[{"name":"Division of Cell Matrix Biology & Regenerative Medicine, School of Biological Sciences, Faculty of Biology, Medicine and Health, University of Manchester, Manchester M13 9PT, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shoko","family":"Ishibashi","sequence":"additional","affiliation":[{"name":"Division of Cell Matrix Biology & Regenerative Medicine, School of Biological Sciences, Faculty of Biology, Medicine and Health, University of Manchester, Manchester M13 9PT, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1805-8548","authenticated-orcid":false,"given":"Enrique","family":"Amaya","sequence":"additional","affiliation":[{"name":"Division of Cell Matrix Biology & Regenerative Medicine, School of Biological Sciences, Faculty of Biology, Medicine and Health, University of Manchester, Manchester M13 9PT, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2019,1,1]]},"reference":[{"key":"2021042500015239300_BIO037655C1","doi-asserted-by":"crossref","first-page":"1872","DOI":"10.1002\/dvdy.20831","article-title":"Analysis of origin and growth of the thyroid gland in zebrafish","volume":"235","author":"Alt","year":"2006","journal-title":"Dev. 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Exp. Zool."},{"key":"2021042500015239300_BIO037655C34","doi-asserted-by":"crossref","first-page":"55","DOI":"10.1007\/s10164-010-0221-5","article-title":"Sex recognition in zebrafish (Danio rerio)","volume":"29","author":"Hutter","year":"2011","journal-title":"J. Ethol."},{"key":"2021042500015239300_BIO037655C35","doi-asserted-by":"crossref","first-page":"1208","DOI":"10.1111\/eth.12027","article-title":"Ephemeral Sexual Dichromatism in Zebrafish (Danio rerio)","volume":"118","author":"Hutter","year":"2012","journal-title":"Ethology"},{"key":"2021042500015239300_BIO037655C36","doi-asserted-by":"crossref","first-page":"450","DOI":"10.1016\/0016-6480(85)90080-2","article-title":"Thyroid hormone induces metamorphosis of flounder larvae","volume":"60","author":"Inui","year":"1985","journal-title":"Gen. Comp. Endocrinol."},{"key":"2021042500015239300_BIO037655C37","doi-asserted-by":"crossref","first-page":"252","DOI":"10.1159\/000362235","article-title":"High frequency of DUOX2 mutations in transient or permanent congenital hypothyroidism with eutopic thyroid glands","volume":"82","author":"Jin","year":"2014","journal-title":"Hormone Res. Paediatr."},{"key":"2021042500015239300_BIO037655C38","doi-asserted-by":"crossref","first-page":"301","DOI":"10.1016\/S0091-679X(08)61831-2","article-title":"Growth control in the ontogenetic and regenerating zebrafish fin","volume":"59","author":"Johnson","year":"1999","journal-title":"Methods Cell Biol."},{"key":"2021042500015239300_BIO037655C39","doi-asserted-by":"crossref","first-page":"1593","DOI":"10.1210\/me.2007-0085","article-title":"Congenital hypothyroidism, dwarfism, and hearing impairment caused by a missense mutation in the mouse dual Oxidase 2 gene, Duox2","volume":"21","author":"Johnson","year":"2007","journal-title":"Mol. 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Engl. J. Med."},{"key":"2021042500015239300_BIO037655C52","doi-asserted-by":"crossref","first-page":"225","DOI":"10.1016\/j.beem.2017.04.006","article-title":"Disorders of H2O2 generation","volume":"31","author":"Muzza","year":"2017","journal-title":"Best Pract. Res. Clin. Endocrinol. Metab."},{"key":"2021042500015239300_BIO037655C53","doi-asserted-by":"crossref","first-page":"145","DOI":"10.1111\/j.1440-169X.2007.00917.x","article-title":"Cellular and molecular processes of regeneration, with special emphasis on fish fins","volume":"49","author":"Nakatani","year":"2007","journal-title":"Dev. Growth Differ."},{"key":"2021042500015239300_BIO037655C54","doi-asserted-by":"crossref","first-page":"192","DOI":"10.1016\/j.aquatox.2015.12.024","article-title":"Impaired anterior swim bladder inflation following exposure to the thyroid peroxidase inhibitor 2-mercaptobenzothiazole part I: fathead minnow","volume":"173","author":"Nelson","year":"2016","journal-title":"Aquat. 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Pediatr."},{"key":"2021042500015239300_BIO037655C64","doi-asserted-by":"crossref","first-page":"376243","DOI":"10.4061\/2011\/376243","article-title":"Alterations along the hypothalamic-pituitary-thyroid axis of the zebrafish (Danio rerio) after exposure to propylthiouracil","volume":"2011","author":"Schmidt","year":"2011","journal-title":"J. Thyroid Res."},{"key":"2021042500015239300_BIO037655C65","doi-asserted-by":"crossref","first-page":"156","DOI":"10.1006\/gcen.1998.7095","article-title":"Metamorphosis in the summer flounder (Paralichthys dentatus): stage-specific developmental response to altered thyroid status","volume":"111","author":"Schreiber","year":"1998","journal-title":"Gen. Comp. 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