{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,7,30]],"date-time":"2025-07-30T09:47:31Z","timestamp":1753868851756,"version":"3.41.2"},"reference-count":23,"publisher":"Wiley","issue":"3-4","license":[{"start":{"date-parts":[[2023,4,14]],"date-time":"2023-04-14T00:00:00Z","timestamp":1681430400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/100000009","name":"Foundation for the National Institutes of Health","doi-asserted-by":"publisher","award":["R01HD105944"],"award-info":[{"award-number":["R01HD105944"]}],"id":[{"id":"10.13039\/100000009","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Genesis"],"published-print":{"date-parts":[[2023,7]]},"abstract":"<jats:title>Summary<\/jats:title><jats:p>Mouse sperm\u2010associated antigen 6 like (SPAG6L) is an axoneme central apparatus protein, essential for the normal function of the ependymal cell and lung cilia, and sperm flagella. Accumulated evidence has disclosed multiple biological functions of SPAG6L, including ciliary\/flagellar biogenesis and polarization, neurogenesis, and neuronal migration. Conventional <jats:italic>Spag6l<\/jats:italic> knockout mice died of hydrocephalus, which impedes further investigation of the function of the gene in vivo. To overcome the limitation of the short lifespan of conventional knockout mice, we developed a conditional allele by inserting two loxP sites in the genome flanking exon 3 of the <jats:italic>Spag6l<\/jats:italic> gene. By crossing the floxed <jats:italic>Spag6l<\/jats:italic> mice to a <jats:italic>Hrpt\u2010Cre<\/jats:italic> line which expresses Cre recombinase ubiquitously in vivo, mutant mice that are missing SPAG6L globally were obtained. Homozygous mutant <jats:italic>Spag6l<\/jats:italic> mice showed normal appearance within the first week after birth, but reduced body size was observed after 1\u00a0week, and all developed hydrocephalus and died within 4\u2009weeks of age. The phenotype mirrored that of the conventional <jats:italic>Spag6l<\/jats:italic> knockout mice. The newly established floxed <jats:italic>Spag6l<\/jats:italic> model provides a powerful tool to further investigate the role of the <jats:italic>Spag6l<\/jats:italic> gene in individual cell types and tissues.<\/jats:p>","DOI":"10.1002\/dvg.23512","type":"journal-article","created":{"date-parts":[[2023,4,14]],"date-time":"2023-04-14T15:38:58Z","timestamp":1681486738000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Generation of floxed <scp><i>Spag6l<\/i><\/scp> mice and disruption of the gene by crossing to a <scp><i>Hprt\u2010Cre<\/i><\/scp> line"],"prefix":"10.1002","volume":"61","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-2730-8649","authenticated-orcid":false,"given":"Yonghong","family":"Man","sequence":"first","affiliation":[{"name":"Department of Occupational and Environmental Health, School of Public Health Wuhan University of Science and Technology  Wuhan Hubei China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Wei","family":"Li","sequence":"additional","affiliation":[{"name":"Department of Physiology Wayne State University  Detroit Michigan USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yi Tian","family":"Yap","sequence":"additional","affiliation":[{"name":"Department of Physiology Wayne State University  Detroit Michigan USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Alivia","family":"Kearney","sequence":"additional","affiliation":[{"name":"Department of Physiology Wayne State University  Detroit Michigan USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Siu\u2010Pok","family":"Yee","sequence":"additional","affiliation":[{"name":"Health Center University of Connecticut  Storrs Connecticut USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"suffix":"III","given":"Jerome F.","family":"Strauss","sequence":"additional","affiliation":[{"name":"Center for Research on Reproduction and Women's Health, Department of Obstetrics and Gynecology, Perelman School of Medicine University of Pennsylvania  Philadelphia Pennsylvania USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Pamela","family":"Harding","sequence":"additional","affiliation":[{"name":"Hypertension and Vascular Research Division Henry Ford Health System  Detroit Michigan USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shizheng","family":"Song","sequence":"additional","affiliation":[{"name":"Department of Occupational and Environmental Health, School of Public Health Wuhan University of Science and Technology  Wuhan Hubei China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ling","family":"Zhang","sequence":"additional","affiliation":[{"name":"Department of Occupational and Environmental Health, School of Public Health Wuhan University of Science and Technology  Wuhan Hubei 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