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Defects in craniofacial development result in common hereditary disorders. Among genetic factors regulating craniofacial development, dysregulated hedgehog signaling is associated with craniofacial skeletal defects such as orofacial clefting and holoprosencephaly. Here, we characterize craniofacial phenotypes associated with two hedgehog signaling co\u2010receptors,\n                      <jats:italic>cdon<\/jats:italic>\n                      and\n                      <jats:italic>boc<\/jats:italic>\n                      , in zebrafish. Genetic pedigree analyses have previously linked both\n                      <jats:italic>cdon<\/jats:italic>\n                      and\n                      <jats:italic>boc<\/jats:italic>\n                      to microform holoprosencephaly, and mutations of Cdon and Boc in mouse result in craniofacial phenotypes. However, a detailed analysis of craniofacial phenotypes associated with\n                      <jats:italic>cdon<\/jats:italic>\n                      and\n                      <jats:italic>boc<\/jats:italic>\n                      in zebrafish has not been completed.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      Our studies show that\n                      <jats:italic>cdon<\/jats:italic>\n                      and\n                      <jats:italic>boc act<\/jats:italic>\n                      redundantly to promote craniofacial cartilage, tendon, and muscle development in zebrafish. Using RNA\u2010seq and HCR in situ hybridization, we show that mutations of\n                      <jats:italic>cdon<\/jats:italic>\n                      and\n                      <jats:italic>boc<\/jats:italic>\n                      result in misregulation of chondrogenesis gene expression including\n                      <jats:italic>Indian hedgehog ligand<\/jats:italic>\n                      , tendon\u2010associated thrombospondin genes, and FOX transcription factors.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>\n                      Our data are consistent with a model whereby\n                      <jats:italic>cdon<\/jats:italic>\n                      and\n                      <jats:italic>boc<\/jats:italic>\n                      together modify hedgehog activity in the head and establish a foundation for using zebrafish to further understand the role of\n                      <jats:italic>cdon<\/jats:italic>\n                      and\n                      <jats:italic>boc<\/jats:italic>\n                      in craniofacial hereditary disorders such as holoprosencephaly.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/dvdy.70153","type":"journal-article","created":{"date-parts":[[2026,5,30]],"date-time":"2026-05-30T10:57:26Z","timestamp":1780138646000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["The hedgehog co\u2010receptors\n                    <i>cdon<\/i>\n                    and\n                    <i>boc<\/i>\n                    function redundantly to regulate zebrafish craniofacial development"],"prefix":"10.1002","author":[{"given":"Ryan","family":"Nickens","sequence":"first","affiliation":[{"name":"Department of Biology Lafayette College  Easton Pennsylvania 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