{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,9]],"date-time":"2026-07-09T20:13:02Z","timestamp":1783627982238,"version":"3.55.0"},"reference-count":75,"publisher":"Oxford University Press (OUP)","issue":"14","license":[{"start":{"date-parts":[[2026,7,9]],"date-time":"2026-07-09T00:00:00Z","timestamp":1783555200000},"content-version":"vor","delay-in-days":189,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100000024","name":"Canadian Institutes of Health Research","doi-asserted-by":"publisher","award":["PJT-166182"],"award-info":[{"award-number":["PJT-166182"]}],"id":[{"id":"10.13039\/501100000024","id-type":"DOI","asserted-by":"publisher"}]},{"name":"CRC Tier 1 chair","award":["#CRC-2021-00441"],"award-info":[{"award-number":["#CRC-2021-00441"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,6,6]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Robinow Syndrome is a genetically heterogeneous, rare skeletal disorder characterized by craniofacial and limb defects. All 7 causative genes lie in the Wingless-related Integration site-1 (WNT) pathway. Here we study the pathogenesis of DVL1 (Dishevelled 1), the most commonly affected gene, where variants cause a frameshift that replaces the C terminus with a novel peptide. We compared phenotypes caused by DVL11519\u0394T to the effects of wtDVL1 or DVL1 with a stop codon at position 1519. Misexpression of DVL11519\u0394T in chicken embryos with an avian retrovirus, leads to increased width of the frontonasal mass similar to the facial phenotype in RS. Ultimately skeletogenesis is inhibited, which was verified in primary cultures of frontonasal mass mesenchyme. In luciferase assays carried out in facial mesenchyme, wtDVL1 activated canonical and JNK-PCP WNT signalling whereas the DVL11519* and the DVL11519\u0394T variant had significantly lower signaling activity. These data confirm that the C-terminus plays an important role in WNT signal transduction and skeletogenesis. We also determined that there is mislocalization of the protein expressed from DVL11519\u0394T in the nucleus while the other two constructs were expressed in the cytoplasm. Nuclear expression of DVL1 may alter transcription in RS. In complementary Drosophila experiments using a variety of readouts, only the DVL11519\u0394T variant and not the 1519* impacted morphogenesis and signaling. This is the first study to show that the novel C-terminus of DVL1 is sufficient to interfere with the function of DVL1 protein expressed from the normal allele in heterozygous, autosomal dominant RS.<\/jats:p>","DOI":"10.1093\/hmg\/ddag053","type":"journal-article","created":{"date-parts":[[2026,7,9]],"date-time":"2026-07-09T19:15:37Z","timestamp":1783624537000},"source":"Crossref","is-referenced-by-count":0,"title":["The abnormal C-terminus caused by\n                    <i>DVL1<\/i>\n                    variants impacts Robinow syndrome phenotypes"],"prefix":"10.1093","volume":"35","author":[{"given":"Shruti S","family":"Tophkhane","sequence":"first","affiliation":[{"name":"Life Sciences Institute and Faculty of Dentistry, University of British Columbia , 2350 Health Sciences Mall Vancouver , BC, V6T 1Z3,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gamze","family":"Akarsu","sequence":"additional","affiliation":[{"name":"Department of Molecular Biology and Biochemistry, Centre for Cell Biology, Development and Disease, Simon Fraser University , 8888 University Drive, Burnaby , BC, V5A 1S6,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sarah J","family":"Gignac","sequence":"additional","affiliation":[{"name":"Life Sciences Institute and Faculty of Dentistry, University of British Columbia , 2350 Health Sciences Mall Vancouver , BC, V6T 1Z3,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xinyi","family":"Xie","sequence":"additional","affiliation":[{"name":"Life Sciences Institute and Faculty of Dentistry, University of British Columbia , 2350 Health Sciences Mall Vancouver , BC, V6T 1Z3,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katherine","family":"Fu","sequence":"additional","affiliation":[{"name":"Life Sciences Institute and Faculty of Dentistry, University of British Columbia , 2350 Health Sciences Mall Vancouver , BC, V6T 1Z3,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9795-5094","authenticated-orcid":false,"given":"Esther M","family":"Verheyen","sequence":"additional","affiliation":[{"name":"Department of Molecular Biology and Biochemistry, Centre for Cell Biology, Development and Disease, Simon Fraser University , 8888 University Drive, Burnaby , BC, V5A 1S6,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1409-8163","authenticated-orcid":false,"given":"Joy M","family":"Richman","sequence":"additional","affiliation":[{"name":"Life Sciences Institute and Faculty of Dentistry, University of British Columbia , 2350 Health Sciences Mall Vancouver , BC, V6T 1Z3,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2026,7,9]]},"reference":[{"key":"2026070915152978100_ref1","doi-asserted-by":"publisher","first-page":"100074","DOI":"10.1016\/j.xhgg.2021.100074","article-title":"Novel pathogenic variants and quantitative phenotypic analyses of Robinow syndrome: WNT signaling perturbation and phenotypic variability","volume":"3","author":"Zhang","year":"2022","journal-title":"HGG Adv"},{"key":"2026070915152978100_ref2","doi-asserted-by":"publisher","first-page":"423","DOI":"10.1038\/78113","article-title":"Mutation of the gene encoding the ROR2 tyrosine kinase causes autosomal recessive Robinow syndrome","volume":"25","author":"Bokhoven","year":"2000","journal-title":"Nat Genet"},{"key":"2026070915152978100_ref3","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1002\/humu.10233","article-title":"One gene, two phenotypes: ROR2 mutations in autosomal recessive Robinow syndrome and autosomal dominant brachydactyly type B","volume":"22","author":"Afzal","year":"2003","journal-title":"Hum Mutat"},{"key":"2026070915152978100_ref4","doi-asserted-by":"publisher","first-page":"27","DOI":"10.1016\/j.ajhg.2017.10.002","article-title":"WNT Signaling perturbations underlie the genetic heterogeneity of Robinow syndrome","volume":"102","author":"White","year":"2018","journal-title":"Am J Hum Genet"},{"key":"2026070915152978100_ref5","doi-asserted-by":"publisher","first-page":"59","DOI":"10.1016\/bs.ctdb.2022.02.008","article-title":"Different strategies by distinct Wnt-signaling pathways in activating a nuclear transcriptional response","volume":"149","author":"Vuong","year":"2022","journal-title":"Curr Top Dev Biol"},{"key":"2026070915152978100_ref6","doi-asserted-by":"publisher","first-page":"75","DOI":"10.1016\/bs.ctdb.2015.11.027","article-title":"The dishevelled protein family: still rather a mystery after over 20 years of molecular studies","volume":"117","author":"Mlodzik","year":"2016","journal-title":"Curr Top Dev Biol"},{"key":"2026070915152978100_ref7","doi-asserted-by":"publisher","first-page":"985","DOI":"10.1016\/j.cell.2017.05.016","article-title":"Wnt\/beta-catenin Signaling, disease, and emerging therapeutic modalities","volume":"169","author":"Nusse","year":"2017","journal-title":"Cell"},{"key":"2026070915152978100_ref8","doi-asserted-by":"publisher","first-page":"2395","DOI":"10.1093\/hmg\/ddz071","article-title":"Robinow syndrome skeletal phenotypes caused by the WNT5AC83S variant are due to dominant interference with chondrogenesis","volume":"28","author":"Gignac","year":"2019","journal-title":"Hum Mol Genet"},{"key":"2026070915152978100_ref9","doi-asserted-by":"publisher","DOI":"10.1242\/dmm.049844","article-title":"Mechanistic studies in drosophila and chicken give new insights into functions of DVL1 in dominant Robinow syndrome","volume":"16","author":"Gignac","year":"2023","journal-title":"Dis Model Mech"},{"key":"2026070915152978100_ref10","doi-asserted-by":"publisher","DOI":"10.1242\/dmm.050584","article-title":"Craniofacial studies in chicken embryos confirm the pathogenicity of human FZD2 variants associated with Robinow syndrome","volume":"17","author":"Tophkhane","year":"2024","journal-title":"Dis Model Mech"},{"key":"2026070915152978100_ref11","doi-asserted-by":"publisher","first-page":"3606","DOI":"10.1002\/ajmg.a.61986","article-title":"Craniofacial phenotypes associated with Robinow syndrome","volume":"185","author":"Conlon","year":"2021","journal-title":"Am J Med Genet"},{"key":"2026070915152978100_ref12","doi-asserted-by":"publisher","first-page":"3584","DOI":"10.1002\/ajmg.a.61884","article-title":"Extremity anomalies associated with Robinow syndrome","volume":"185","author":"Abu-Ghname","year":"2021","journal-title":"Am J Med Genet"},{"key":"2026070915152978100_ref13","doi-asserted-by":"publisher","first-page":"2005","DOI":"10.1002\/ajmg.a.61756","article-title":"50 years of Robinow syndrome","volume":"182","author":"Mazzeu","year":"2020","journal-title":"Am J Med Genet"},{"key":"2026070915152978100_ref14","doi-asserted-by":"publisher","first-page":"2632","DOI":"10.1002\/ajmg.a.61843","article-title":"Characterization of the Robinow syndrome skeletal phenotype, bone micro-architecture, and genotype-phenotype correlations with the osteosclerotic form","volume":"182","author":"Shayota","year":"2020","journal-title":"Am J Med Genet"},{"key":"2026070915152978100_ref15","doi-asserted-by":"publisher","first-page":"623","DOI":"10.1016\/j.ajhg.2015.02.010","article-title":"Mutations in DVL1 cause an osteosclerotic form of Robinow syndrome","volume":"96","author":"Bunn","year":"2015","journal-title":"Am J Hum Genet"},{"key":"2026070915152978100_ref16","doi-asserted-by":"publisher","first-page":"612","DOI":"10.1016\/j.ajhg.2015.02.015","article-title":"DVL1 frameshift mutations clustering in the penultimate exon cause autosomal-dominant Robinow syndrome","volume":"96","author":"White","year":"2015","journal-title":"Am J Hum Genet"},{"key":"2026070915152978100_ref17","doi-asserted-by":"publisher","first-page":"553","DOI":"10.1016\/j.ajhg.2016.01.005","article-title":"DVL3 alleles resulting in a \u22121 frameshift of the last exon mediate autosomal-dominant Robinow syndrome","volume":"98","author":"White","year":"2016","journal-title":"Am J Hum Genet"},{"key":"2026070915152978100_ref18","doi-asserted-by":"publisher","first-page":"717","DOI":"10.1016\/j.cellsig.2009.11.021","article-title":"Dishevelled: the hub of Wnt signaling","volume":"22","author":"Gao","year":"2010","journal-title":"Cell Signal"},{"key":"2026070915152978100_ref19","doi-asserted-by":"publisher","first-page":"e1886","DOI":"10.1002\/mgg3.1886","article-title":"A novel frameshift mutation of DVL1-induced Robinow syndrome: a case report and literature review","volume":"10","author":"Hu","year":"2022","journal-title":"Mol Genet Genomic Med"},{"key":"2026070915152978100_ref20","doi-asserted-by":"publisher","first-page":"73","DOI":"10.1006\/dbio.1994.1297","article-title":"Isolation and characterization of a mouse homolog of the drosophila segment polarity gene dishevelled","volume":"166","author":"Sussman","year":"1994","journal-title":"Dev Biol"},{"key":"2026070915152978100_ref21","doi-asserted-by":"publisher","first-page":"2044","DOI":"10.1002\/dvdy.22028","article-title":"Diversification of the expression patterns and developmental functions of the dishevelled gene family during chordate evolution","volume":"238","author":"Gray","year":"2009","journal-title":"Dev Dyn"},{"key":"2026070915152978100_ref22","doi-asserted-by":"publisher","first-page":"52","DOI":"10.1016\/j.cellsig.2018.03.004","article-title":"Dishevelled: a masterful conductor of complex Wnt signals","volume":"47","author":"Sharma","year":"2018","journal-title":"Cell Signal"},{"key":"2026070915152978100_ref23","doi-asserted-by":"publisher","first-page":"5898","DOI":"10.1074\/jbc.M116.772509","article-title":"Autoinhibition of dishevelled protein regulated by its extreme C terminus plays a distinct role in Wnt\/beta-catenin and Wnt\/planar cell polarity (PCP) signaling pathways","volume":"292","author":"Qi","year":"2017","journal-title":"J Biol Chem"},{"key":"2026070915152978100_ref24","doi-asserted-by":"publisher","first-page":"1087","DOI":"10.1083\/jcb.200710050","article-title":"Nuclear Dvl, c-Jun, beta-catenin, and TCF form a complex leading to stabilization of beta-catenin-TCF interaction","volume":"180","author":"Gan","year":"2008","journal-title":"J Cell Biol"},{"key":"2026070915152978100_ref25","doi-asserted-by":"publisher","first-page":"3","DOI":"10.1186\/jbiol20","article-title":"Nuclear localization is required for dishevelled function in Wnt\/beta-catenin signaling","volume":"4","author":"Itoh","year":"2005","journal-title":"J Biol"},{"key":"2026070915152978100_ref26","doi-asserted-by":"publisher","first-page":"4421","DOI":"10.1242\/dev.02068","article-title":"The developmental biology of dishevelled: an enigmatic protein governing cell fate and cell polarity","volume":"132","author":"Wallingford","year":"2005","journal-title":"Development"},{"key":"2026070915152978100_ref27","doi-asserted-by":"publisher","first-page":"119305","DOI":"10.1016\/j.bbamcr.2022.119305","article-title":"Nuclear dishevelled: an enigmatic role in governing cell fate and Wnt signaling","volume":"1869","author":"Boligala","year":"2022","journal-title":"Biochim Biophys Acta Mol Cell Res"},{"key":"2026070915152978100_ref28","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1186\/jbiol21","article-title":"Dishevelled nuclear shuttling","volume":"4","author":"Weitzman","year":"2005","journal-title":"J Biol"},{"key":"2026070915152978100_ref29","doi-asserted-by":"publisher","first-page":"4414","DOI":"10.1128\/MCB.19.6.4414","article-title":"DIX domains of Dvl and axin are necessary for protein interactions and their ability to regulate beta-catenin stability","volume":"19","author":"Kishida","year":"1999","journal-title":"Mol Cell Biol"},{"key":"2026070915152978100_ref30","doi-asserted-by":"publisher","first-page":"1010","DOI":"10.1093\/emboj\/19.5.1010","article-title":"Dishevelled phosphorylation, subcellular localization and multimerization regulate its role in early embryogenesis","volume":"19","author":"Rothbacher","year":"2000","journal-title":"EMBO J"},{"key":"2026070915152978100_ref31","doi-asserted-by":"publisher","first-page":"1211","DOI":"10.1242\/dev.126.6.1211","article-title":"A Wnt5a pathway underlies outgrowth of multiple structures in the vertebrate embryo","volume":"126","author":"Yamaguchi","year":"1999","journal-title":"Development"},{"key":"2026070915152978100_ref32","doi-asserted-by":"publisher","first-page":"213","DOI":"10.1016\/B978-0-12-394592-1.00007-7","article-title":"Dishevelled: in vivo roles of a multifunctional gene family during development","volume":"101","author":"Wynshaw-Boris","year":"2012","journal-title":"Curr Top Dev Biol"},{"key":"2026070915152978100_ref33","doi-asserted-by":"publisher","first-page":"3707","DOI":"10.1242\/dev.052001","article-title":"Frizzled 1 and frizzled 2 genes function in palate, ventricular septum and neural tube closure: general implications for tissue fusion processes","volume":"137","author":"Yu","year":"2010","journal-title":"Development"},{"key":"2026070915152978100_ref34","doi-asserted-by":"publisher","first-page":"895","DOI":"10.1016\/S0092-8674(00)80354-2","article-title":"Social interaction and sensorimotor gating abnormalities in mice lacking Dvl1","volume":"90","author":"Lijam","year":"1997","journal-title":"Cell"},{"key":"2026070915152978100_ref35","doi-asserted-by":"publisher","first-page":"5827","DOI":"10.1242\/dev.00164","article-title":"Dishevelled 2 is essential for cardiac outflow tract development, somite segmentation and neural tube closure","volume":"129","author":"Hamblet","year":"2002","journal-title":"Development"},{"key":"2026070915152978100_ref36","doi-asserted-by":"publisher","first-page":"e1000259","DOI":"10.1371\/journal.pgen.1000259","article-title":"Murine dishevelled 3 functions in redundant pathways with dishevelled 1 and 2 in normal cardiac outflow tract, cochlea, and neural tube development","volume":"4","author":"Etheridge","year":"2008","journal-title":"PLoS Genet"},{"key":"2026070915152978100_ref37","doi-asserted-by":"publisher","first-page":"1265","DOI":"10.1177\/0022034517716916","article-title":"Abnormal WNT5A Signaling causes mandibular hypoplasia in Robinow syndrome","volume":"96","author":"Hosseini-Farahabadi","year":"2017","journal-title":"J Dent Res"},{"key":"2026070915152978100_ref38","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1002\/dvdy.70056","article-title":"Robinow syndrome DVL1 variants disrupt morphogenesis and appendage formation in a drosophila disease model","author":"Akarsu","year":"2025","journal-title":"Dev Dyn TBD"},{"key":"2026070915152978100_ref39","doi-asserted-by":"publisher","first-page":"252","DOI":"10.1016\/j.matbio.2013.02.005","article-title":"Dual functions for WNT5A during cartilage development and in disease","volume":"32","author":"Hosseini-Farahabadi","year":"2013","journal-title":"Matrix Biol"},{"key":"2026070915152978100_ref40","doi-asserted-by":"publisher","first-page":"2233","DOI":"10.3174\/ajnr.A3415","article-title":"Illustrated review of the embryology and development of the facial region, part 1: early face and lateral nasal cavities","volume":"34","author":"Som","year":"2013","journal-title":"AJNR Am J Neuroradiol"},{"key":"2026070915152978100_ref41","doi-asserted-by":"publisher","first-page":"209","DOI":"10.1083\/jcb.123.1.209","article-title":"Tissue polarity genes of drosophila regulate the subcellular location for prehair initiation in pupal wing cells","volume":"123","author":"Wong","year":"1993","journal-title":"J Cell Biol"},{"key":"2026070915152978100_ref42","doi-asserted-by":"publisher","first-page":"517","DOI":"10.1146\/annurev.genet.42.110807.091432","article-title":"Planar cell polarity signaling: from fly development to human disease","volume":"42","author":"Simons","year":"2008","journal-title":"Annu Rev Genet"},{"key":"2026070915152978100_ref43","doi-asserted-by":"publisher","first-page":"dev193755","DOI":"10.1242\/dev.193755","article-title":"Symmetry and fluctuation of cell movements in neural crest-derived facial mesenchyme","volume":"148","author":"Danescu","year":"2021","journal-title":"Development"},{"key":"2026070915152978100_ref44","doi-asserted-by":"publisher","first-page":"680","DOI":"10.1016\/S0960-9822(03)00240-9","article-title":"Zebrafish prickle, a modulator of noncanonical Wnt\/Fz signaling, regulates gastrulation movements","volume":"13","author":"Veeman","year":"2003","journal-title":"Curr Biol"},{"key":"2026070915152978100_ref45","doi-asserted-by":"publisher","first-page":"188","DOI":"10.1002\/dvdy.22500","article-title":"An ATF2-based luciferase reporter to monitor non-canonical Wnt signaling in Xenopus embryos","volume":"240","author":"Ohkawara","year":"2011","journal-title":"Dev Dyn"},{"key":"2026070915152978100_ref46","doi-asserted-by":"publisher","first-page":"2610","DOI":"10.1101\/gad.12.16.2610","article-title":"Differential recruitment of dishevelled provides signaling specificity in the planar cell polarity and wingless signaling pathways","volume":"12","author":"Axelrod","year":"1998","journal-title":"Genes Dev"},{"key":"2026070915152978100_ref47","doi-asserted-by":"publisher","first-page":"1883","DOI":"10.1242\/dev.120.7.1883","article-title":"A single frizzled protein has a dual function in tissue polarity","volume":"120","author":"Krasnow","year":"1994","journal-title":"Development"},{"key":"2026070915152978100_ref48","doi-asserted-by":"publisher","first-page":"549","DOI":"10.1038\/329549a0","article-title":"Directional non-cell autonomy and the transmission of polarity information by the frizzled gene of drosophila","volume":"329","author":"Vinson","year":"1987","journal-title":"Nature"},{"key":"2026070915152978100_ref49","doi-asserted-by":"publisher","first-page":"4725","DOI":"10.1242\/dev.01369","article-title":"The involvement of Frodo in TCF-dependent signaling and neural tissue development","volume":"131","author":"Hikasa","year":"2004","journal-title":"Development"},{"key":"2026070915152978100_ref50","doi-asserted-by":"publisher","first-page":"351","DOI":"10.1038\/ncb784","article-title":"Frodo interacts with dishevelled to transduce Wnt signals","volume":"4","author":"Gloy","year":"2002","journal-title":"Nat Cell Biol"},{"key":"2026070915152978100_ref51","doi-asserted-by":"publisher","first-page":"e60865","DOI":"10.1371\/journal.pone.0060865","article-title":"IQGAP1 functions as a modulator of dishevelled nuclear localization in Wnt signaling","volume":"8","author":"Goto","year":"2013","journal-title":"PLoS One"},{"key":"2026070915152978100_ref52","doi-asserted-by":"publisher","first-page":"1804","DOI":"10.1038\/s41467-019-09651-7","article-title":"Dishevelled-3 conformation dynamics analyzed by FRET-based biosensors reveals a key role of casein kinase 1","volume":"10","author":"Harnos","year":"2019","journal-title":"Nat Commun"},{"key":"2026070915152978100_ref53","doi-asserted-by":"publisher","first-page":"8388","DOI":"10.1038\/s41598-022-10536-x","article-title":"DVL1 and DVL3 require nuclear localisation to regulate proliferation in human myoblasts","volume":"12","author":"Pruller","year":"2022","journal-title":"Sci Rep"},{"key":"2026070915152978100_ref54","doi-asserted-by":"publisher","first-page":"9304","DOI":"10.1073\/pnas.1608783113","article-title":"Dishevelled is a NEK2 kinase substrate controlling dynamics of centrosomal linker proteins","volume":"113","author":"Cervenka","year":"2016","journal-title":"Proc Natl Acad Sci U S A"},{"key":"2026070915152978100_ref55","doi-asserted-by":"publisher","first-page":"15","DOI":"10.1111\/j.1399-0004.2011.01683.x","article-title":"Craniofacial and intraoral phenotype of Robinow syndrome forms","volume":"80","author":"Beiraghi","year":"2011","journal-title":"Clin Genet"},{"key":"2026070915152978100_ref56","doi-asserted-by":"publisher","first-page":"843","DOI":"10.1016\/S0092-8674(01)00614-6","article-title":"Wnt\/frizzled activation of rho regulates vertebrate gastrulation and requires a novel Formin homology protein Daam1","volume":"107","author":"Habas","year":"2001","journal-title":"Cell"},{"key":"2026070915152978100_ref57","first-page":"21","article-title":"Functional cranial analysis and the functional matrix","volume":"17","author":"Moss","year":"1979","journal-title":"Int J Orthod"},{"key":"2026070915152978100_ref58","doi-asserted-by":"publisher","first-page":"1446","DOI":"10.1101\/gad.990702","article-title":"FGF signaling pathways in endochondral and intramembranous bone development and human genetic disease","volume":"16","author":"Ornitz","year":"2002","journal-title":"Genes Dev"},{"key":"2026070915152978100_ref59","doi-asserted-by":"publisher","first-page":"643","DOI":"10.1016\/j.devcel.2016.08.011","article-title":"Generating cellular diversity and spatial form: Wnt Signaling and the evolution of multicellular animals","volume":"38","author":"Loh","year":"2016","journal-title":"Dev Cell"},{"key":"2026070915152978100_ref60","doi-asserted-by":"publisher","first-page":"364","DOI":"10.1038\/nsmb912","article-title":"Beta-catenin directly displaces Groucho\/TLE repressors from Tcf\/Lef in Wnt-mediated transcription activation","volume":"12","author":"Daniels","year":"2005","journal-title":"Nat Struct Mol Biol"},{"key":"2026070915152978100_ref61","doi-asserted-by":"publisher","DOI":"10.3390\/cells13221870","article-title":"Structural and functional insights into dishevelled-mediated Wnt Signaling","volume":"13","author":"Wang","year":"2024","journal-title":"Cells"},{"key":"2026070915152978100_ref62","doi-asserted-by":"crossref","DOI":"10.64898\/2026.02.14.705933","article-title":"The abnormal C-terminus in DVL1 impacts Robinow syndrome phenotypes","author":"Tophkhane","year":"2026"},{"key":"2026070915152978100_ref63","doi-asserted-by":"publisher","first-page":"707","DOI":"10.1016\/j.devcel.2015.01.031","article-title":"FOXKs promote Wnt\/beta-catenin signaling by translocating DVL into the nucleus","volume":"32","author":"Wang","year":"2015","journal-title":"Dev Cell"},{"key":"2026070915152978100_ref64","doi-asserted-by":"publisher","first-page":"6288","DOI":"10.1038\/s41467-025-61551-1","article-title":"Dishevelled-1 regulates global transcriptomic changes and associates with ETS1 transcription factor","volume":"16","author":"Martinez-Marin","year":"2025","journal-title":"Nat Commun"},{"key":"2026070915152978100_ref65","doi-asserted-by":"publisher","first-page":"59","DOI":"10.3389\/fcell.2017.00059","article-title":"Dishevelled paralogs in vertebrate development: redundant or distinct?","volume":"5","author":"Gentzel","year":"2017","journal-title":"Front Cell Dev Biol"},{"key":"2026070915152978100_ref66","doi-asserted-by":"publisher","first-page":"231","DOI":"10.1002\/aja.1001950404","article-title":"A series of normal stages in the development of the chick embryo","volume":"195","author":"Hamburger","year":"1992","journal-title":"Dev Dyn"},{"key":"2026070915152978100_ref67","doi-asserted-by":"publisher","first-page":"221","DOI":"10.1093\/dnares\/8.5.221","article-title":"Generation of RCAS vectors useful for functional genomic analyses","volume":"8","author":"Loftus","year":"2001","journal-title":"DNA Res"},{"key":"2026070915152978100_ref68","doi-asserted-by":"publisher","first-page":"107","DOI":"10.14712\/fb2004050030107","article-title":"The RCAS vector system","volume":"50","author":"Hughes","year":"2004","journal-title":"Folia Biol (Praha)"},{"key":"2026070915152978100_ref69","doi-asserted-by":"publisher","first-page":"24153","DOI":"10.1074\/jbc.M113.522003","article-title":"Avian facial morphogenesis is regulated by c-Jun N-terminal kinase\/planar cell polarity (JNK\/PCP) wingless-related (WNT) Signaling","volume":"289","author":"Geetha-Loganathan","year":"2014","journal-title":"J Biol Chem"},{"key":"2026070915152978100_ref70","doi-asserted-by":"publisher","first-page":"4428","DOI":"10.1242\/dev.081679","article-title":"Twist1 mediates repression of chondrogenesis by \u03b2-catenin to promote cranial bone progenitor specification","volume":"139","author":"Goodnough","year":"2012","journal-title":"Development"},{"key":"2026070915152978100_ref71","doi-asserted-by":"publisher","first-page":"49","DOI":"10.1002\/jmor.1050880104","article-title":"A series of normal stages in the development of the chick embryo","volume":"88","author":"Hamburger","year":"1951","journal-title":"J Morphol"},{"key":"2026070915152978100_ref72","doi-asserted-by":"publisher","first-page":"251","DOI":"10.1007\/978-1-62703-989-5_19","article-title":"Analysis of chondrogenesis using micromass cultures of limb mesenchyme","volume":"1130","author":"Underhill","year":"2014","journal-title":"Methods Mol Biol"},{"key":"2026070915152978100_ref73","doi-asserted-by":"publisher","first-page":"1101","DOI":"10.1038\/nprot.2008.73","article-title":"Analyzing real-time PCR data by the comparative CT method","volume":"3","author":"Schmittgen","year":"2008","journal-title":"Nat Protoc"},{"key":"2026070915152978100_ref74","doi-asserted-by":"publisher","first-page":"676","DOI":"10.1038\/nmeth.2019","article-title":"Fiji: an open-source platform for biological-image analysis","volume":"9","author":"Schindelin","year":"2012","journal-title":"Nat Methods"},{"key":"2026070915152978100_ref75","doi-asserted-by":"publisher","first-page":"671","DOI":"10.1038\/nmeth.2089","article-title":"NIH image to ImageJ: 25 years of image analysis","volume":"9","author":"Schneider","year":"2012","journal-title":"Nat Methods"}],"container-title":["Human Molecular Genetics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/hmg\/article-pdf\/35\/14\/ddag053\/68741002\/ddag053.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/hmg\/article-pdf\/35\/14\/ddag053\/68741002\/ddag053.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,7,9]],"date-time":"2026-07-09T19:15:39Z","timestamp":1783624539000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/hmg\/article\/doi\/10.1093\/hmg\/ddag053\/8729340"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026]]},"references-count":75,"journal-issue":{"issue":"14","published-print":{"date-parts":[[2026,6,6]]}},"URL":"https:\/\/doi.org\/10.1093\/hmg\/ddag053","relation":{},"ISSN":["0964-6906","1460-2083"],"issn-type":[{"value":"0964-6906","type":"print"},{"value":"1460-2083","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2026]]},"published":{"date-parts":[[2026]]},"article-number":"ddag053"}}