{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,22]],"date-time":"2026-05-22T05:06:44Z","timestamp":1779426404000,"version":"3.53.1"},"reference-count":46,"publisher":"Wiley","license":[{"start":{"date-parts":[[2026,5,21]],"date-time":"2026-05-21T00:00:00Z","timestamp":1779321600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,5,21]],"date-time":"2026-05-21T00:00:00Z","timestamp":1779321600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/501100000274","name":"British Heart Foundation","doi-asserted-by":"publisher","award":["RG\/19\/2\/34256"],"award-info":[{"award-number":["RG\/19\/2\/34256"]}],"id":[{"id":"10.13039\/501100000274","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000274","name":"British Heart Foundation","doi-asserted-by":"publisher","award":["FS\/IBSRF\/24\/25222"],"award-info":[{"award-number":["FS\/IBSRF\/24\/25222"]}],"id":[{"id":"10.13039\/501100000274","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["anatomypubs.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Developmental Dynamics"],"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>\n                      Human mutations in\n                      <jats:italic>INVERSIN<\/jats:italic>\n                      are associated with nephronophthisis, variable penetrance of\n                      <jats:italic>situs inversus<\/jats:italic>\n                      and congenital heart disease. Inversin has been shown to localize to cilia and many of the patient phenotypes are attributed to disrupted cilia function. We set out to characterize the\n                      <jats:italic>invs<\/jats:italic>\n                      <jats:sup>\n                        <jats:italic>sa12246<\/jats:italic>\n                      <\/jats:sup>\n                      allele in zebrafish to investigate its utility in understanding patient disease.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      The left\u2013right axis is established correctly in\n                      <jats:italic>invs<\/jats:italic>\n                      <jats:sup>\n                        <jats:italic>sa12246<\/jats:italic>\n                      <\/jats:sup>\n                      mutants, kidneys appear to develop normally without any cysts and cilia appear normal, however mutants are significantly shorter. In post\u2010embryonic stages,\n                      <jats:italic>invs<\/jats:italic>\n                      <jats:sup>\n                        <jats:italic>sa12246<\/jats:italic>\n                      <\/jats:sup>\n                      mutants display significant growth delay and signs of retinal mis\u2010patterning together with spinal deformities reminiscent of idiopathic scoliosis. The allele is lethal in the juvenile stage.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>\n                      We show that\n                      <jats:italic>invs<\/jats:italic>\n                      <jats:sup>\n                        <jats:italic>sa12246<\/jats:italic>\n                      <\/jats:sup>\n                      allele has a distinct phenotype from other models where\n                      <jats:italic>inversin<\/jats:italic>\n                      function is disrupted, uncovering novel roles in post\u2010embryonic development.\n                    <\/jats:p>\n                  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