{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,21]],"date-time":"2026-09-21T00:06:48Z","timestamp":1789949208076,"version":"4.0.1"},"reference-count":25,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2007,1,10]],"date-time":"2007-01-10T00:00:00Z","timestamp":1168387200000},"content-version":"vor","delay-in-days":9,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Genesis"],"published-print":{"date-parts":[[2007,1]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p><jats:italic>SALL4<\/jats:italic> is a homologue of the Drosophila homeotic gene <jats:italic>spalt<\/jats:italic>, a zinc finger transcription factor, required for inner cell mass proliferation in early embryonic development. It also interacts with other transcription factors to control the development of the anorectal region, kidney, heart, limbs, and brain. Truncating mutations in <jats:italic>SALL4<\/jats:italic> cause Okihiro syndrome, manifest as Duane anomaly, radial ray defects and sensorineural and conductive deafness. We report the characterization of a novel murine <jats:italic>Sall4<\/jats:italic> null allele created by bacterial recombineering in ES cells. Homozygous mutant mice exhibit early embryonic lethality. Heterozygous mutant mice recapitulate phenotypic features of Okihiro syndrome including deafness, lower anogenital tract abnormalities, renal hypoplasia, anencephaly, Hirschprung's disease, and skeletal defects. This phenotype shows important differences in cardiac and ear manifestations to previously characterized <jats:italic>Sall4<\/jats:italic> mutant alleles and should prove useful for the investigation of the influence of modifier alleles and protein interactions on the transcriptional regulatory function of <jats:italic>Sall4<\/jats:italic>. genesis 45:51\u201358, 2007. \u00a9 2007 Wiley\u2010Liss, Inc.<\/jats:p>","DOI":"10.1002\/dvg.20264","type":"journal-article","created":{"date-parts":[[2007,1,10]],"date-time":"2007-01-10T19:18:47Z","timestamp":1168456727000},"page":"51-58","source":"Crossref","is-referenced-by-count":62,"title":["A <i>Sall4<\/i> mutant mouse model useful for studying the role of <i>Sall4<\/i> in early embryonic development and 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