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Disruptions in the structure and function of primary cilia cause inherited disorders called ciliopathies. We aimed to evaluate Sprouty2 and Sprouty4 gene-dependent alterations of ciliary structure and to focus on the determination of its association with Hedgehog signaling defects in chondrocytes. Analysis of the transgenic mice phenotype with Sprouty2 and Sprouty4 deficiency revealed several defects, including improper endochondral bone formation and digit patterning, or craniofacial and dental abnormalities. Moreover, reduced bone thickness and trabecular bone mass, skull deformities, or chondroma-like lesions were revealed. All these pathologies might be attributed to ciliopathies. Elongation of the ciliary axonemes in embryonic and postnatal growth plate chondrocytes was observed in Sprouty2\u2212\/\u2212 and Sprouty2+\/\u2212\/Sprouty4\u2212\/\u2212 mutants compared with corresponding littermate controls. Also, cilia-dependent Hedgehog signaling was upregulated in Sprouty2\/4 mutant animals. Ptch1 and Ihh expression were upregulated in the autopodium and the proximal tibia of Sprouty2\u2212\/\u2212\/Sprouty4\u2212\/\u2212 mutants. Increased levels of the GLI3 repressor (GLI3R) form were detected in Sprouty2\/4 mutant primary fibroblast embryonic cell cultures and tissues. These findings demonstrate that mouse lines deficient in Sprouty proteins manifest phenotypic features resembling ciliopathic phenotypes in multiple aspects and may serve as valuable models to study the association between overactivation of RTK and dysfunction of primary cilia during skeletogenesis. \u00a9 2021 American Society for Bone and Mineral Research (ASBMR).<\/jats:p>","DOI":"10.1002\/jbmr.4427","type":"journal-article","created":{"date-parts":[[2021,8,23]],"date-time":"2021-08-23T06:55:10Z","timestamp":1629701710000},"page":"2258-2274","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":9,"title":["Loss of Sprouty Produces a Ciliopathic Skeletal Phenotype in Mice Through Upregulation of Hedgehog Signaling"],"prefix":"10.1093","volume":"36","author":[{"given":"Eva","family":"Hruba","sequence":"first","affiliation":[{"name":"Department of Experimental Biology, Faculty of Science Masaryk University Brno Czech Republic"},{"name":"Institute of Animal Physiology and Genetics Czech Academy of Sciences Brno Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michaela","family":"Kavkova","sequence":"additional","affiliation":[{"name":"Central European Institute of Technology Brno University of Technology Brno Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0512-2122","authenticated-orcid":false,"given":"Linda","family":"Dalecka","sequence":"additional","affiliation":[{"name":"Department of Cell Biology, Faculty of Science Charles University Prague Czech Republic"},{"name":"Institute of Histology and Embryology, First Faculty of Medicine Charles University Prague Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5663-6831","authenticated-orcid":false,"given":"Milo\u0161","family":"Macholan","sequence":"additional","affiliation":[{"name":"Institute of Animal Physiology and Genetics Czech Academy of Sciences Brno Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tomas","family":"Zikmund","sequence":"additional","affiliation":[{"name":"Central European Institute of Technology Brno University of Technology Brno Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Miroslav","family":"Varecha","sequence":"additional","affiliation":[{"name":"Department of Biology, Faculty of Medicine Masaryk University Brno Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7627-0344","authenticated-orcid":false,"given":"Michaela","family":"Bosakova","sequence":"additional","affiliation":[{"name":"Department of Biology, Faculty of Medicine Masaryk University Brno Czech Republic"},{"name":"Institute of Animal Physiology and Genetics Czech Academy of Sciences Brno Czech 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Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0262-6774","authenticated-orcid":false,"given":"Marcela","family":"Buchtova","sequence":"additional","affiliation":[{"name":"Department of Experimental Biology, Faculty of Science Masaryk University Brno Czech Republic"},{"name":"Institute of Animal Physiology and Genetics Czech Academy of Sciences Brno Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2021,11,1]]},"reference":[{"issue":"2","key":"2024022211011615100_jbmr4427-bib-0001","doi-asserted-by":"crossref","first-page":"253","DOI":"10.1016\/S0092-8674(00)80919-8","article-title":"Sprouty encodes a novel antagonist of FGF signaling that patterns apical branching of the Drosophila 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