{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,10]],"date-time":"2026-10-10T04:22:47Z","timestamp":1791606167231,"version":"4.3.4"},"reference-count":61,"publisher":"The Company of Biologists","issue":"7","license":[{"start":{"date-parts":[[2026,6,26]],"date-time":"2026-06-26T00:00:00Z","timestamp":1782432000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"name":"National Institute of Health","award":["K08DE031039"],"award-info":[{"award-number":["K08DE031039"]}]},{"DOI":"10.13039\/100000072","name":"National Institute of Dental and Craniofacial Research","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100000072","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100006019","name":"Van Andel Research Institute","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100006019","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,7,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>Wnt receptors of the Frizzled (Fzd) family are widely considered to exhibit substantial functional redundancy, complicating efforts to therapeutically target individual receptors. Fzd2 was believed to be functionally redundant with Fzd1 and Fzd7, based on previously published global knockout mouse studies. By contrast, homozygosity for a Fzd2 global knockout mouse allele developed by the International Mouse Phenotype Consortium (IMPC) has been reported to cause embryonic lethality, suggesting that Fzd2 is critical for early embryonic development. If global deletion of Fzd2 leads to early lethality, conditional deletion models are necessary to identify tissue-specific phenotypes. We found that a previously published Fzd2 conditional deletion model does not eliminate Fzd2. We have generated a new conditional model to address the contradictory previous studies and allow tissue-specific studies of Fzd2. We successfully inserted two loxP sites around the Fzd2 gene and confirmed that subsequent Cre-mediated recombination creates a Fzd2 null allele. Global deletion of Fzd2 in this model does not cause embryonic lethality while limb-specific deletion causes limb shortening. This work supports the hypothesis that Fzd2 regulates limb development and emphasizes the importance of thoroughly validating newly generated mouse models.<\/jats:p>","DOI":"10.1242\/dmm.052410","type":"journal-article","created":{"date-parts":[[2026,6,15]],"date-time":"2026-06-15T07:12:41Z","timestamp":1781507561000},"source":"Crossref","is-referenced-by-count":1,"title":["Clarifying Frizzled 2 function in development through genetically validated mouse models"],"prefix":"10.1242","volume":"19","author":[{"given":"Megan N.","family":"Michalski","sequence":"first","affiliation":[{"name":"Van Andel Institute 1 Department of Cell Biology , , Grand Rapids, Michigan 49503 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cassandra R.","family":"Diegel","sequence":"additional","affiliation":[{"name":"Van Andel Institute 1 Department of Cell Biology , , Grand Rapids, Michigan 49503 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zhendong A.","family":"Zhong","sequence":"additional","affiliation":[{"name":"Van Andel Institute 1 Department of Cell Biology , , Grand Rapids, Michigan 49503 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maddison E.","family":"Marshall","sequence":"additional","affiliation":[{"name":"Van Andel Institute 1 Department of Cell Biology , , Grand Rapids, Michigan 49503 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gabrielle E.","family":"Foxa Wiartalla","sequence":"additional","affiliation":[{"name":"Van Andel Institute 1 Department of Cell Biology , , Grand Rapids, Michigan 49503 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Payton D.","family":"Stevens","sequence":"additional","affiliation":[{"name":"Van Andel Institute 1 Department of Cell Biology , , Grand Rapids, Michigan 49503 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kelly","family":"Suino-Powell","sequence":"additional","affiliation":[{"name":"Van Andel Institute 2 Department of Structural Biology , , Grand Rapids, Michigan 49503 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Levi L.","family":"Blazer","sequence":"additional","affiliation":[{"name":"University of Toronto 3 Donnelly Centre for Cellular and Biomolecular Research , , Toronto, Ontario M5S 3E1 ,","place":["Canada"]},{"name":"School of Pharmacy, University of Waterloo 4 , Kitchener, Ontario N2G 1C5 ,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jarrett J.","family":"Adams","sequence":"additional","affiliation":[{"name":"University of Toronto 3 Donnelly Centre for Cellular and Biomolecular Research , , Toronto, Ontario M5S 3E1 ,","place":["Canada"]},{"name":"School of Pharmacy, University of Waterloo 4 , Kitchener, Ontario N2G 1C5 ,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Karsten","family":"Melcher","sequence":"additional","affiliation":[{"name":"Van Andel Institute 2 Department of Structural Biology , , Grand Rapids, Michigan 49503 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sachdev S.","family":"Sidhu","sequence":"additional","affiliation":[{"name":"University of Toronto 3 Donnelly Centre for Cellular and Biomolecular Research , , Toronto, Ontario M5S 3E1 ,","place":["Canada"]},{"name":"School of Pharmacy, University of Waterloo 4 , Kitchener, Ontario N2G 1C5 ,","place":["Canada"]},{"name":"University of Toronto 5 Department of Molecular Genetics , , Toronto, Ontario M5S 3G9 ,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stephane","family":"Angers","sequence":"additional","affiliation":[{"name":"University of Toronto 3 Donnelly Centre for Cellular and Biomolecular Research , , Toronto, Ontario M5S 3E1 ,","place":["Canada"]},{"name":"University of Toronto 6 Department of Pharmaceutical Sciences, Leslie Dan Faculty of Pharmacy , , Toronto, Ontario M5S 3M2 ,","place":["Canada"]},{"name":"University of Toronto 7 Department of Biochemistry , , Toronto, Ontario M5S 1A8 ,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5261-5301","authenticated-orcid":false,"given":"Bart O.","family":"Williams","sequence":"additional","affiliation":[{"name":"Van Andel Institute 1 Department of Cell Biology , , Grand Rapids, Michigan 49503 ,","place":["USA"]},{"name":"Van Andel Institute 8 Core Technologies and Services , , Grand Rapids, Michigan 49503 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2026,6,26]]},"reference":[{"key":"2026072404012796600_DMM052410C1","doi-asserted-by":"publisher","first-page":"419","DOI":"10.1038\/78107","article-title":"Recessive Robinow syndrome, allelic to dominant brachydactyly type B, is caused by mutation of ROR2","volume":"25","author":"Afzal","year":"2000","journal-title":"Nat. Genet."},{"key":"2026072404012796600_DMM052410C59","doi-asserted-by":"publisher","first-page":"2260","DOI":"10.1016\/j.jid.2022.01.004","article-title":"FZD2 Regulates Murine Hair Follicle Function and Maintenance","volume":"142","author":"Brennan-Crispi","year":"2022","journal-title":"J Invest Dermatol"},{"key":"2026072404012796600_DMM052410C2","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":"2026072404012796600_DMM052410C60","doi-asserted-by":"publisher","first-page":"242","DOI":"10.1093\/nar\/gky354","article-title":"CRISPOR: intuitive guide selection for CRISPR\/Cas9 genome editing experiments and screens","volume":"46","author":"Concordet","year":"2018","journal-title":"Nucleic Acids Res."},{"key":"2026072404012796600_DMM052410C3","doi-asserted-by":"publisher","first-page":"113","DOI":"10.1016\/j.trecan.2017.01.001","article-title":"Wnt\/planar cell polarity signaling: new opportunities for cancer treatment","volume":"3","author":"Daulat","year":"2017","journal-title":"Trends Cancer"},{"key":"2026072404012796600_DMM052410C5","doi-asserted-by":"publisher","first-page":"305","DOI":"10.1101\/gad.235473.113","article-title":"The R-spondin\/Lgr5\/Rnf43 module: regulator of Wnt signal strength","volume":"28","author":"de Lau","year":"2014","journal-title":"Genes Dev."},{"key":"2026072404012796600_DMM052410C6","doi-asserted-by":"publisher","first-page":"262","DOI":"10.1016\/j.ymeth.2009.12.007","article-title":"Accurate and objective copy number profiling using real-time quantitative PCR","volume":"50","author":"D'Haene","year":"2010","journal-title":"Methods"},{"key":"2026072404012796600_DMM052410C7","doi-asserted-by":"publisher","first-page":"508","DOI":"10.1038\/nature19356","article-title":"High-throughput discovery of novel developmental phenotypes","volume":"537","author":"Dickinson","year":"2016","journal-title":"Nature"},{"key":"2026072404012796600_DMM052410C8","doi-asserted-by":"publisher","first-page":"e1008940","DOI":"10.1371\/journal.pgen.1008940","article-title":"Independent validation of experimental results requires timely and unrestricted access to animal models and reagents","volume":"16","author":"Diegel","year":"2020","journal-title":"PLoS Genet."},{"key":"2026072404012796600_DMM052410C9","doi-asserted-by":"publisher","first-page":"eaat1178","DOI":"10.1126\/science.aat1178","article-title":"A molecular mechanism for Wnt ligand-specific signaling","volume":"361","author":"Eubelen","year":"2018","journal-title":"Science"},{"key":"2026072404012796600_DMM052410C10","doi-asserted-by":"publisher","first-page":"1323","DOI":"10.1016\/j.mri.2012.05.001","article-title":"3D Slicer as an image computing platform for the Quantitative Imaging Network","volume":"30","author":"Fedorov","year":"2012","journal-title":"Magn. Reson. Imaging"},{"key":"2026072404012796600_DMM052410C11","doi-asserted-by":"publisher","first-page":"dmm049844","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":"2026072404012796600_DMM052410C12","doi-asserted-by":"publisher","first-page":"632","DOI":"10.1038\/nbt.4166","article-title":"Efficient generation of targeted large insertions by microinjection into two-cell-stage mouse embryos","volume":"36","author":"Gu","year":"2018","journal-title":"Nat. Biotechnol."},{"key":"2026072404012796600_DMM052410C13","doi-asserted-by":"publisher","first-page":"2452","DOI":"10.1038\/s41596-019-0187-x","article-title":"Creation of CRISPR-based germline-genome-engineered mice without ex vivo handling of zygotes by i-GONAD","volume":"14","author":"Gurumurthy","year":"2019","journal-title":"Nat. Protoc."},{"key":"2026072404012796600_DMM052410C14","doi-asserted-by":"publisher","first-page":"15.7.1","DOI":"10.1002\/0471142905.hg1507s83","article-title":"Mouse genome editing using the CRISPR\/Cas system","volume":"83","author":"Harms","year":"2014","journal-title":"Curr. Protoc. Hum. Genet."},{"key":"2026072404012796600_DMM052410C15","doi-asserted-by":"publisher","first-page":"437","DOI":"10.1016\/S1535-6108(03)00309-X","article-title":"Preinvasive and invasive ductal pancreatic cancer and its early detection in the mouse","volume":"4","author":"Hingorani","year":"2003","journal-title":"Cancer Cell"},{"key":"2026072404012796600_DMM052410C16","doi-asserted-by":"publisher","first-page":"469","DOI":"10.1016\/j.ccr.2005.04.023","article-title":"Trp53R172H and KrasG12D cooperate to promote chromosomal instability and widely metastatic pancreatic ductal adenocarcinoma in mice","volume":"7","author":"Hingorani","year":"2005","journal-title":"Cancer Cell"},{"key":"2026072404012796600_DMM052410C17","doi-asserted-by":"publisher","first-page":"e3133","DOI":"10.21769\/BioProtoc.3133","article-title":"Cartilage induction from mouse mesenchymal stem cells in high-density micromass culture","volume":"9","author":"Iezaki","year":"2019","journal-title":"Bio Protoc."},{"key":"2026072404012796600_DMM052410C18","doi-asserted-by":"publisher","first-page":"12444","DOI":"10.1073\/pnas.1406639111","article-title":"Wnt ligand\/+ 2 receptor signaling regulates tube shape and branch-point formation in the lung through control of epithelial cell shape","volume":"111","author":"Kadzik","year":"2014","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2026072404012796600_DMM052410C19","doi-asserted-by":"publisher","first-page":"8125","DOI":"10.1093\/nar\/15.20.8125","article-title":"An analysis of 5'-noncoding sequences from 699 vertebrate messenger RNAs","volume":"15","author":"Kozak","year":"1987","journal-title":"Nucleic Acids Res."},{"key":"2026072404012796600_DMM052410C20","doi-asserted-by":"publisher","first-page":"229","DOI":"10.1016\/j.ydbio.2013.04.026","article-title":"The widely used Wnt1-Cre transgene causes developmental phenotypes by ectopic activation of Wnt signaling","volume":"379","author":"Lewis","year":"2013","journal-title":"Dev. Biol."},{"key":"2026072404012796600_DMM052410C62","doi-asserted-by":"publisher","first-page":"3094","DOI":"10.1093\/bioinformatics\/bty191","article-title":"Minimap2: pairwise alignment for nucleotide sequences","volume":"34","author":"Li","year":"2018","journal-title":"Bioinformatics"},{"key":"2026072404012796600_DMM052410C61","doi-asserted-by":"publisher","first-page":"1754","DOI":"10.1093\/bioinformatics\/btp324","article-title":"Fast and accurate short read alignment with Burrows-Wheeler transform","volume":"25","author":"Li","year":"2009","journal-title":"Bioinformatics"},{"key":"2026072404012796600_DMM052410C21","doi-asserted-by":"publisher","first-page":"dev201038","DOI":"10.1242\/dev.201038","article-title":"Successful therapeutic intervention in new mouse models of frizzled 2-associated congenital malformations","volume":"150","author":"Liegel","year":"2023","journal-title":"Development"},{"key":"2026072404012796600_DMM052410C22","doi-asserted-by":"publisher","first-page":"77","DOI":"10.1002\/gene.10092","article-title":"Expression of Cre Recombinase in the developing mouse limb bud driven by a Prxl enhancer","volume":"33","author":"Logan","year":"2002","journal-title":"Genesis"},{"key":"2026072404012796600_DMM052410C23","doi-asserted-by":"publisher","first-page":"823","DOI":"10.1126\/science.1232033","article-title":"RNA-guided human genome engineering via Cas9","volume":"339","author":"Mali","year":"2013","journal-title":"Science"},{"key":"2026072404012796600_DMM052410C24","doi-asserted-by":"publisher","first-page":"347","DOI":"10.1038\/nature03837","article-title":"Neural crest origins of the neck and shoulder","volume":"436","author":"Matsuoka","year":"2005","journal-title":"Nature"},{"key":"2026072404012796600_DMM052410C25","doi-asserted-by":"publisher","first-page":"371","DOI":"10.1038\/s41580-024-00823-y","article-title":"Mechanistic insights into Wnt-","volume":"26","author":"Maurice","year":"2025","journal-title":"Nat. Rev. Mol. Cell Biol."},{"key":"2026072404012796600_DMM052410C26","first-page":"259","article-title":"In vitro pre-validation of gene editing by CRISPR\/Cas9 ribonucleoprotein","volume":"11","author":"Mehravar","year":"2019","journal-title":"Avicenna J. Med. Biotechnol."},{"key":"2026072404012796600_DMM052410C27","doi-asserted-by":"publisher","DOI":"10.1101\/2021.01.27.428341","article-title":"Generation of a new frizzled 2 flox mouse model to clarify its role in development","author":"Michalski","year":"2021","journal-title":"bioRxiv"},{"key":"2026072404012796600_DMM052410C28","doi-asserted-by":"publisher","first-page":"7461","DOI":"10.1038\/sj.onc.1210053","article-title":"Wnts as ligands: processing, secretion and reception","volume":"25","author":"Mikels","year":"2006","journal-title":"Oncogene"},{"key":"2026072404012796600_DMM052410C29","doi-asserted-by":"publisher","first-page":"eaay4494","DOI":"10.1126\/scisignal.aay4494","article-title":"Perspectives on the role of Wnt biology in cancer","volume":"12","author":"Mirabelli","year":"2019","journal-title":"Sci. Signal."},{"key":"2026072404012796600_DMM052410C30","doi-asserted-by":"publisher","first-page":"982","DOI":"10.1038\/nmeth.3543","article-title":"CRISPRscan: designing highly efficient sgRNAs for CRISPR-Cas9 targeting in vivo","volume":"12","author":"Moreno-Mateos","year":"2015","journal-title":"Nat. Methods"},{"key":"2026072404012796600_DMM052410C31","doi-asserted-by":"publisher","first-page":"739","DOI":"10.1002\/ajmg.a.38623","article-title":"Nonsense mutations in FZD2 cause autosomal-dominant omodysplasia: Robinow syndrome-like phenotypes","volume":"176","author":"Nagasaki","year":"2018","journal-title":"Am. J. Med. Genet. A"},{"key":"2026072404012796600_DMM052410C32","doi-asserted-by":"publisher","first-page":"985","DOI":"10.1016\/j.cell.2017.05.016","article-title":"Wnt","volume":"169","author":"Nusse","year":"2017","journal-title":"Cell"},{"key":"2026072404012796600_DMM052410C33","doi-asserted-by":"publisher","first-page":"1124","DOI":"10.1016\/j.stemcr.2017.08.007","article-title":"Ontogenic identification and analysis of mesenchymal stromal cell populations during mouse limb and long bone development","volume":"9","author":"Nusspaumer","year":"2017","journal-title":"Stem Cell Rep."},{"key":"2026072404012796600_DMM052410C34","doi-asserted-by":"publisher","first-page":"25","DOI":"10.1186\/s13059-018-1400-x","article-title":"i-GONAD: a robust method for in situ germline genome engineering using CRISPR nucleases","volume":"19","author":"Ohtsuka","year":"2018","journal-title":"Genome Biol."},{"key":"2026072404012796600_DMM052410C35","doi-asserted-by":"publisher","first-page":"847","DOI":"10.1016\/j.cell.2004.11.004","article-title":"Mutant p53 gain of function in two mouse models of Li-Fraumeni syndrome","volume":"119","author":"Olive","year":"2004","journal-title":"Cell"},{"key":"2026072404012796600_DMM052410C36","doi-asserted-by":"publisher","first-page":"327","DOI":"10.1002\/dvdy.22156","article-title":"WNT5A mutations in patients with autosomal dominant Robinow syndrome","volume":"239","author":"Person","year":"2010","journal-title":"Dev. Dyn."},{"key":"2026072404012796600_DMM052410C37","doi-asserted-by":"publisher","first-page":"dev173328","DOI":"10.1242\/dev.173328","article-title":"Molecular signatures identify immature mesenchymal progenitors in early mouse limb buds that respond differentially to morphogen signaling","volume":"146","author":"Reinhardt","year":"2019","journal-title":"Development"},{"key":"2026072404012796600_DMM052410C38","doi-asserted-by":"publisher","first-page":"217","DOI":"10.4161\/org.7.3.18583","article-title":"Cell polarity: the missing link in skeletal morphogenesis?","volume":"7","author":"Romereim","year":"2011","journal-title":"Organogenesis"},{"key":"2026072404012796600_DMM052410C39","doi-asserted-by":"publisher","first-page":"3399","DOI":"10.1093\/hmg\/ddv088","article-title":"A mutation in FRIZZLED2 impairs Wnt signaling and causes autosomal dominant omodysplasia","volume":"24","author":"Saal","year":"2015","journal-title":"Hum. Mol. Genet."},{"key":"2026072404012796600_DMM052410C40","doi-asserted-by":"publisher","first-page":"e33761","DOI":"10.7554\/eLife.33761","article-title":"Covalent linkage of the DNA repair template to the CRISPR-Cas9 nuclease enhances homology-directed repair","volume":"7","author":"Savic","year":"2018","journal-title":"eLife"},{"key":"2026072404012796600_DMM052410C41","doi-asserted-by":"publisher","first-page":"e3136","DOI":"10.21769\/BioProtoc.3136","article-title":"In vitro generation of CRISPR-Cas9 complexes with covalently bound repair templates for genome editing in mammalian cells","volume":"9","author":"Savi\u0107","year":"2019","journal-title":"Bio. Protoc."},{"key":"2026072404012796600_DMM052410C42","doi-asserted-by":"publisher","first-page":"5080","DOI":"10.1093\/nar\/23.24.5080","article-title":"A cre-transgenic mouse strain for the ubiquitous deletion of loxP-flanked gene segments including deletion in germ cells","volume":"23","author":"Schwenk","year":"1995","journal-title":"Nucleic Acids Res."},{"key":"2026072404012796600_DMM052410C43","doi-asserted-by":"publisher","first-page":"e46134","DOI":"10.7554\/eLife.46134","article-title":"Tailored tetravalent antibodies potently and specifically activate Wnt\/Frizzled pathways in cells, organoids and mice","volume":"8","author":"Tao","year":"2019","journal-title":"eLife"},{"key":"2026072404012796600_DMM052410C44","doi-asserted-by":"publisher","first-page":"dmm050584","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":"2026072404012796600_DMM052410C45","doi-asserted-by":"publisher","first-page":"318","DOI":"10.1159\/000479721","article-title":"A novel de novo FZD2 mutation in a patient with autosomal dominant omodysplasia","volume":"8","author":"T\u00fcrkmen","year":"2017","journal-title":"Mol. Syndromol."},{"key":"2026072404012796600_DMM052410C46","doi-asserted-by":"publisher","DOI":"10.1007\/978-1-62703-989-5_19","article-title":"Analysis of chondrogenesis using micromass cultures of limb mesenchyme","volume-title":"Skeletal Development and Repair: Methods and Protocols","author":"Underhill","year":"2014"},{"key":"2026072404012796600_DMM052410C47","doi-asserted-by":"publisher","first-page":"367","DOI":"10.1016\/S1534-5807(03)00266-1","article-title":"A second canon. Functions and mechanisms of beta-catenin-independent Wnt signaling","volume":"5","author":"Veeman","year":"2003","journal-title":"Dev. Cell"},{"key":"2026072404012796600_DMM052410C48","doi-asserted-by":"publisher","first-page":"113","DOI":"10.1016\/bs.ctdb.2015.11.028","article-title":"Frizzled receptors in development and disease","volume":"117","author":"Wang","year":"2016","journal-title":"Curr. Top. Dev. Biol."},{"key":"2026072404012796600_DMM052410C49","doi-asserted-by":"publisher","first-page":"2252","DOI":"10.1002\/ccr3.1818","article-title":"Two unrelated patients with autosomal dominant omodysplasia and FRIZZLED2 mutations","volume":"6","author":"Warren","year":"2018","journal-title":"Clin. Case Rep."},{"key":"2026072404012796600_DMM052410C50","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":"2026072404012796600_DMM052410C51","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":"2026072404012796600_DMM052410C52","doi-asserted-by":"publisher","first-page":"623","DOI":"10.1146\/annurev-cellbio-100814-125315","article-title":"Wnt-Frizzled\/planar cell polarity signaling: cellular orientation by facing the wind (Wnt)","volume":"31","author":"Yang","year":"2015","journal-title":"Annu. Rev. Cell Dev. Biol."},{"key":"2026072404012796600_DMM052410C53","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":"2026072404012796600_DMM052410C54","doi-asserted-by":"publisher","first-page":"4383","DOI":"10.1242\/dev.083352","article-title":"Frizzled 2 and frizzled 7 function redundantly in convergent extension and closure of the ventricular septum and palate: evidence for a network of interacting genes","volume":"139","author":"Yu","year":"2012","journal-title":"Development"},{"key":"2026072404012796600_DMM052410C55","doi-asserted-by":"publisher","first-page":"44005","DOI":"10.1074\/jbc.M208265200","article-title":"Osteoblast-specific knockout of the insulin-like growth factor (IGF) receptor gene reveals an essential role of IGF signaling in bone matrix mineralization","volume":"277","author":"Zhang","year":"2002","journal-title":"J. Biol. Chem."},{"key":"2026072404012796600_DMM052410C56","doi-asserted-by":"publisher","first-page":"119","DOI":"10.1007\/978-1-4939-6393-5_13","article-title":"Use of primary calvarial osteoblasts to evaluate the function of Wnt signaling in osteogenesis","volume":"1481","author":"Zhong","year":"2016","journal-title":"Methods Mol. Biol."},{"key":"2026072404012796600_DMM052410C57","doi-asserted-by":"publisher","first-page":"100782","DOI":"10.1016\/j.jbc.2021.100782","article-title":"Regulation of Wnt receptor activity: Implications for therapeutic development in colon cancer","volume":"296","author":"Zhong","year":"2021","journal-title":"J. Biol. Chem."},{"key":"2026072404012796600_DMM052410C58","doi-asserted-by":"publisher","first-page":"dmm049876","DOI":"10.1242\/dmm.049876","article-title":"FZD2 regulates limb development by mediating \u03b2-catenin-dependent and -independent Wnt signaling pathways","volume":"16","author":"Zhu","year":"2023","journal-title":"Dis. Model. Mech."}],"container-title":["Disease Models &amp; Mechanisms"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.biologists.com\/dmm\/article-pdf\/doi\/10.1242\/dmm.052410\/3704219\/dmm052410.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/journals.biologists.com\/dmm\/article-pdf\/doi\/10.1242\/dmm.052410\/3705784\/dmm052410.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/journals.biologists.com\/dmm\/article-pdf\/doi\/10.1242\/dmm.052410\/3705784\/dmm052410.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,7,24]],"date-time":"2026-07-24T08:01:34Z","timestamp":1784880094000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.biologists.com\/dmm\/article\/19\/7\/dmm052410\/372074\/Clarifying-Frizzled-2-function-in-development"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,6,26]]},"references-count":61,"journal-issue":{"issue":"7","published-print":{"date-parts":[[2026,7,1]]}},"URL":"https:\/\/doi.org\/10.1242\/dmm.052410","relation":{},"ISSN":["1754-8403","1754-8411"],"issn-type":[{"value":"1754-8403","type":"print"},{"value":"1754-8411","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,6,26]]},"article-number":"dmm052410"}}