{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,26]],"date-time":"2026-09-26T05:42:17Z","timestamp":1790401337861,"version":"4.1.0"},"reference-count":30,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2018,10,2]],"date-time":"2018-10-02T00:00:00Z","timestamp":1538438400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100000925","name":"National Health and Medical Research Council","doi-asserted-by":"publisher","award":["APP1084944"],"award-info":[{"award-number":["APP1084944"]}],"id":[{"id":"10.13039\/501100000925","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000925","name":"National Health and Medical Research Council","doi-asserted-by":"publisher","award":["APP1136567"],"award-info":[{"award-number":["APP1136567"]}],"id":[{"id":"10.13039\/501100000925","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000925","name":"National Health and Medical Research Council","doi-asserted-by":"publisher","award":["APP1144159"],"award-info":[{"award-number":["APP1144159"]}],"id":[{"id":"10.13039\/501100000925","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Journal of Anatomy"],"published-print":{"date-parts":[[2018,12]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Klippel\u2013Feil syndrome is a congenital vertebral anomaly, which is characterised by the fusion of at least two cervical vertebrae and a clinically broad set of symptoms, including congenital scoliosis and elevated scapula (Sprengel's deformity). Klippel\u2013Feil syndrome is associated with mutations in <jats:italic><jats:styled-content style=\"fixed-case\">MEOX<\/jats:styled-content>1<\/jats:italic>. The zebrafish mutant <jats:italic>choker<\/jats:italic> (<jats:italic>cho<\/jats:italic>) carries a mutation in its orthologue, <jats:italic>meox1<\/jats:italic>. Although zebrafish is being increasingly employed as fidelitous models of human spinal disease, the vertebral column of Meox1\u2010deficient fish has not been assessed for defects. Here, we describe the skeletal defects of <jats:italic>meox1<\/jats:italic><jats:sup><jats:italic>cho<\/jats:italic><\/jats:sup> mutant zebrafish utilising alizarin red to stain bones and chemical maceration of soft tissue to detect fusions in an unbiased manner. Obtained data reveal that <jats:italic>meox1<\/jats:italic><jats:sup><jats:italic>cho<\/jats:italic><\/jats:sup> mutants feature aspects of a number of described symptoms of patients who suffer from Klippel\u2013Feil syndrome and have mutations in <jats:italic><jats:styled-content style=\"fixed-case\">MEOX<\/jats:styled-content>1<\/jats:italic>. These include vertebral fusion, congenital scoliosis and an asymmetry of the pectoral girdle, which resembles Sprengel's deformity. Thus, the <jats:italic>meox1<\/jats:italic><jats:sup><jats:italic>cho<\/jats:italic><\/jats:sup> mutant zebrafish may serve as a useful tool to study the pathogenesis of the symptoms associated with Klippel\u2013Feil syndrome.<\/jats:p>","DOI":"10.1111\/joa.12890","type":"journal-article","created":{"date-parts":[[2018,10,2]],"date-time":"2018-10-02T11:09:28Z","timestamp":1538478568000},"page":"687-695","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":29,"title":["Skeletal malformations of Meox1\u2010deficient zebrafish resemble human Klippel\u2013Feil syndrome"],"prefix":"10.1111","volume":"233","author":[{"given":"Mervyn V. P.","family":"Dauer","sequence":"first","affiliation":[{"name":"Australian Regenerative Medicine Institute Monash University Clayton VIC Australia"},{"name":"Victoria Node EMBL Australia Clayton VIC Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Peter D.","family":"Currie","sequence":"additional","affiliation":[{"name":"Australian Regenerative Medicine Institute Monash University Clayton VIC Australia"},{"name":"Victoria Node EMBL Australia Clayton VIC Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7859-545X","authenticated-orcid":false,"given":"Joachim","family":"Berger","sequence":"additional","affiliation":[{"name":"Australian Regenerative Medicine Institute Monash University Clayton VIC Australia"},{"name":"Victoria Node EMBL Australia Clayton VIC 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