{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,16]],"date-time":"2026-09-16T16:44:42Z","timestamp":1789577082455,"version":"build-2803163510"},"reference-count":40,"publisher":"SAGE Publications","issue":"1","license":[{"start":{"date-parts":[[2018,3,27]],"date-time":"2018-03-27T00:00:00Z","timestamp":1522108800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["J Hand Surg Eur Vol"],"published-print":{"date-parts":[[2019,1]]},"abstract":"<jats:p>Preaxial polydactyly is a congenital hand anomaly predominantly of sporadic occurrence, which is frequently associated with abnormalities of the Sonic hedgehog signalling pathway. In experimentally induced preaxial polydactyly, radial aplasia is also frequently observed. To determine if there is a correlation between preaxial polydactyly and radial aplasia, we induced ectopic Sonic hedgehog signalling during chicken limb development with application of a smoothened-agonist (SAG) or retinoic acid. Application of SAG caused malformations in 71% limbs including preaxial polydactyly (62%) and forearm abnormalities (43%). Retinoic acid application induced malformations in 56% of limb including preaxial polydactyly (45%) and forearm abnormalities (50%). Radial dysplasia and ulnar dimelia were observed in both experimental conditions. We demonstrate that ectopic Sonic hedgehog signalling may cause both preaxial polydactyly and predictable forearm anomalies and that these conditions could potentially be classified as one embryological group. We propose a unifying model based on known models of ectopic Sonic hedgehog signalling.<\/jats:p>","DOI":"10.1177\/1753193418762959","type":"journal-article","created":{"date-parts":[[2018,3,28]],"date-time":"2018-03-28T01:50:38Z","timestamp":1522201838000},"page":"43-50","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":23,"title":["Experimental evidence that preaxial polydactyly and forearm radial deficiencies may share a common developmental origin"],"prefix":"10.1177","volume":"44","author":[{"given":"Wee L.","family":"Lam","sequence":"first","affiliation":[{"name":"Royal Hospital for Sick Children, Edinburgh, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Julia D. H.","family":"Oh","sequence":"additional","affiliation":[{"name":"Royal Hospital for Sick Children, Edinburgh, UK"},{"name":"Division of Developmental Biology, University of Edinburgh, Edinburgh, UK. In collaboration with Roslin Institute Chicken Embryology (RICE)"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Edward J.","family":"Johnson","sequence":"additional","affiliation":[{"name":"Division of Developmental Biology, University of Edinburgh, Edinburgh, UK. In collaboration with Roslin Institute Chicken Embryology (RICE)"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sandra Poyatos","family":"Pertinez","sequence":"additional","affiliation":[{"name":"Division of Developmental Biology, University of Edinburgh, Edinburgh, UK. In collaboration with Roslin Institute Chicken Embryology (RICE)"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chloe","family":"Stephens","sequence":"additional","affiliation":[{"name":"Division of Developmental Biology, University of Edinburgh, Edinburgh, UK. In collaboration with Roslin Institute Chicken Embryology (RICE)"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Megan G.","family":"Davey","sequence":"additional","affiliation":[{"name":"Division of Developmental Biology, University of Edinburgh, Edinburgh, UK. In collaboration with Roslin Institute Chicken Embryology (RICE)"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2018,3,27]]},"reference":[{"key":"bibr1-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1097\/SAP.0b013e318248b67f"},{"key":"bibr2-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1016\/S0266-7681(98)80140-X"},{"key":"bibr3-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.35584"},{"key":"bibr4-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1016\/j.tig.2012.03.012"},{"key":"bibr5-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pbio.1001957"},{"key":"bibr6-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1016\/j.ydbio.2012.07.017"},{"key":"bibr7-1753193418762959","first-page":"403","volume-title":"Congenital malformation of the hand and forearm","author":"Buck-Gramcko D","year":"1998"},{"key":"bibr8-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1006\/dbio.2001.0346"},{"key":"bibr9-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1101\/gad.369106"},{"key":"bibr10-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1002\/dvdy.22623"},{"key":"bibr11-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1016\/S0925-4773(99)00040-4"},{"key":"bibr12-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.101.5.1913"},{"key":"bibr13-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1038\/nature08117"},{"key":"bibr14-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1002\/aja.1001950404"},{"key":"bibr15-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1016\/j.cell.2004.07.024"},{"key":"bibr16-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.31336"},{"key":"bibr17-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1016\/j.ydbio.2014.05.025"},{"key":"bibr18-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1242\/dev.00861"},{"key":"bibr19-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1002\/aja.1002040411"},{"key":"bibr20-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1016\/j.jhsa.2006.09.010"},{"key":"bibr21-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1016\/j.jhsa.2010.09.031"},{"key":"bibr22-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1006\/dbio.1996.0282"},{"key":"bibr23-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1016\/0092-8674(93)90626-2"},{"key":"bibr24-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1242\/dev.00245"},{"key":"bibr25-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1126\/science.1199489"},{"key":"bibr26-1753193418762959","first-page":"227","volume":"32","author":"Summerbell D","year":"1974","journal-title":"J Embryol Exp Morphol"},{"key":"bibr27-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1038\/289295a0"},{"key":"bibr28-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1038\/254199a0"},{"key":"bibr29-1753193418762959","first-page":"14","volume":"28","author":"Tickle C","year":"2017","journal-title":"Front Cell Dev Biol"},{"key":"bibr30-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1007\/978-1-59745-516-9_2"},{"key":"bibr31-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1038\/nature06718"},{"key":"bibr32-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1038\/ncomms1437"},{"key":"bibr33-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1038\/352539a0"},{"key":"bibr34-1753193418762959","first-page":"175","volume":"64","author":"Wassel HD","year":"1969","journal-title":"Clin Orthop Relat Res"},{"key":"bibr35-1753193418762959","first-page":"125","volume":"1","author":"Wolpert L","year":"1968","journal-title":"Towards a theoretical biology"},{"key":"bibr36-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1016\/S0363-5023(78)80136-1"},{"key":"bibr37-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1242\/dev.124.21.4393"},{"key":"bibr38-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1097\/01.sap.0000171426.12675.dd"},{"key":"bibr39-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1016\/j.devcel.2008.01.008"},{"key":"bibr40-1753193418762959","doi-asserted-by":"publisher","DOI":"10.1016\/j.jhsa.2007.12.012"}],"container-title":["Journal of Hand Surgery (European Volume)"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/1753193418762959","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/full-xml\/10.1177\/1753193418762959","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/1753193418762959","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,4,29]],"date-time":"2026-04-29T22:48:40Z","timestamp":1777502920000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.sagepub.com\/doi\/10.1177\/1753193418762959"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2018,3,27]]},"references-count":40,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2019,1]]}},"alternative-id":["10.1177\/1753193418762959"],"URL":"https:\/\/doi.org\/10.1177\/1753193418762959","relation":{},"ISSN":["1753-1934","2043-6289"],"issn-type":[{"value":"1753-1934","type":"print"},{"value":"2043-6289","type":"electronic"}],"subject":[],"published":{"date-parts":[[2018,3,27]]}}}