{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,7]],"date-time":"2026-10-07T02:52:17Z","timestamp":1791341537943,"version":"4.3.1"},"reference-count":60,"publisher":"The Company of Biologists","issue":"15","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2011,8,1]]},"abstract":"<jats:p>Specification of digit number and identity is central to digit pattern in vertebrate limbs. The classical talpid3 chicken mutant has many unpatterned digits together with defects in other regions, depending on hedgehog (Hh) signalling, and exhibits embryonic lethality. The talpid3 chicken has a mutation in KIAA0586, which encodes a centrosomal protein required for the formation of primary cilia, which are sites of vertebrate Hh signalling. The highly conserved exons 11 and 12 of KIAA0586 are essential to rescue cilia in talpid3 chicken mutants. We constitutively deleted these two exons to make a talpid3\u2013\/\u2013 mouse. Mutant mouse embryos lack primary cilia and, like talpid3 chicken embryos, have face and neural tube defects but also defects in left\/right asymmetry. Conditional deletion in mouse limb mesenchyme results in polydactyly and in brachydactyly and a failure of subperisoteal bone formation, defects that are attributable to abnormal sonic hedgehog and Indian hedgehog signalling, respectively. Like talpid3 chicken limbs, the mutant mouse limbs are syndactylous with uneven digit spacing as reflected in altered Raldh2 expression, which is normally associated with interdigital mesenchyme. Both mouse and chicken mutant limb buds are broad and short. talpid3\u2013\/\u2013 mouse cells migrate more slowly than wild-type mouse cells, a change in cell behaviour that possibly contributes to altered limb bud morphogenesis. This genetic mouse model will facilitate further conditional approaches, epistatic experiments and open up investigation into the function of the novel talpid3 gene using the many resources available for mice.<\/jats:p>","DOI":"10.1242\/dev.063602","type":"journal-article","created":{"date-parts":[[2011,7,12]],"date-time":"2011-07-12T11:32:44Z","timestamp":1310470364000},"page":"3261-3272","source":"Crossref","is-referenced-by-count":78,"title":["Generation of mice with functional inactivation of<i>talpid3<\/i>, a gene first identified in chicken"],"prefix":"10.1242","volume":"138","author":[{"given":"Fiona","family":"Bangs","sequence":"first","affiliation":[{"name":"Biology and Biochemistry Department, University of Bath, Claverton Down, Bath BA2 7AY, UK."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nicole","family":"Antonio","sequence":"additional","affiliation":[{"name":"Biology and Biochemistry Department, University of Bath, Claverton Down, Bath BA2 7AY, UK."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Peerapat","family":"Thongnuek","sequence":"additional","affiliation":[{"name":"Biology and Biochemistry Department, University of Bath, Claverton Down, Bath BA2 7AY, UK."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Monique","family":"Welten","sequence":"additional","affiliation":[{"name":"Biology and Biochemistry Department, University of Bath, Claverton Down, Bath BA2 7AY, UK."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Megan G.","family":"Davey","sequence":"additional","affiliation":[{"name":"Division of Developmental Biology, The Roslin Institute, The University of Edinburgh, Easter Bush, Midlothian, EH25 9RG, Scotland, UK."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"James","family":"Briscoe","sequence":"additional","affiliation":[{"name":"Developmental Neurobiology, National Institute for Medical Research, Mill Hill, London NW7 1AA, UK."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cheryll","family":"Tickle","sequence":"additional","affiliation":[{"name":"Biology and Biochemistry Department, University of Bath, Claverton Down, Bath BA2 7AY, UK."}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","reference":[{"key":"2021042606581405700_R1","first-page":"1185","article-title":"A second talpid-like mutation in the fowl","volume":"38","author":"Abbott","year":"1959","journal-title":"Poult. 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