{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,16]],"date-time":"2026-10-16T17:39:10Z","timestamp":1792172350357,"version":"4.4.0"},"reference-count":46,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2006,8,18]],"date-time":"2006-08-18T00:00:00Z","timestamp":1155859200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Congenital Anomalies"],"published-print":{"date-parts":[[2006,9]]},"abstract":"<jats:p><jats:bold>ABSTRACT\u2003<\/jats:bold>Pattern formation along the body axis directs the proportion of different types of cells required for functional tissue structures. The secreted protein sonic hedgehog (<jats:italic>Shh<\/jats:italic>) and zinc finger transcription factor<jats:italic>Gli3<\/jats:italic>are key players in pattern formation during brain and limb development; the antagonistic action of<jats:italic>Shh<\/jats:italic>towards<jats:italic>Gli3<\/jats:italic>may be crucial for pattern formation. Recent findings from<jats:italic>Shh\/Gli3<\/jats:italic>double homozygous mutants suggest that a balance of both activities is required for the production of the normal proportion of different cell types during organogenesis. This conclusion contrasts with the alternative hypothesis that a<jats:italic>Shh<\/jats:italic>gradient directs the specification of several different cell types. The observations reviewed here offer a new perspective on understanding the pathogenesis of human birth defects caused by mutations of the<jats:italic>Shh<\/jats:italic>and<jats:italic>Gli3<\/jats:italic>genes.<\/jats:p>","DOI":"10.1111\/j.1741-4520.2006.00114.x","type":"journal-article","created":{"date-parts":[[2006,8,18]],"date-time":"2006-08-18T09:53:32Z","timestamp":1155894812000},"page":"123-128","source":"Crossref","is-referenced-by-count":20,"title":["Essential roles of<i>Gli3<\/i>and sonic hedgehog in pattern formation and developmental anomalies caused by their dysfunction"],"prefix":"10.1111","volume":"46","author":[{"given":"Jun","family":"Motoyama","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2006,8,18]]},"reference":[{"key":"e_1_2_6_2_1","doi-asserted-by":"crossref","first-page":"4975","DOI":"10.1242\/dev.129.21.4975","article-title":"Chordin and noggin promote organizing centers 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