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Here, we used the pharmacological inhibitor PLX5622 to disrupt colony stimulating factor 1 receptor (CSF1R) signaling, which is essential for macrophage and osteoclast proliferation, differentiation and survival. Prenatal PLX5622 exposure in mouse resulted in \u223c50% depletion of CSF1R+ macrophages, with complete loss of osteoclasts. While there were no notable changes in craniofacial nerve or muscle development, prenatal exposure to PLX5622 resulted in skull doming and cranial suture impairments, in addition to disruptions to development of the premaxilla, mandible, ear ossicles, palate and cranial base. In response to PLX5622 exposure, cytokine and chemokine signaling was altered and neural crest proliferation was impaired. Our data also highlight sex- and strain-specific differences in PLX5622 phenotypes and together demonstrate that CSF1R+ macrophages and osteoclasts are essential for craniofacial morphogenesis.<\/jats:p>","DOI":"10.1242\/dev.205423","type":"journal-article","created":{"date-parts":[[2026,3,27]],"date-time":"2026-03-27T09:51:58Z","timestamp":1774605118000},"source":"Crossref","is-referenced-by-count":5,"title":["CSF1R+ macrophage and osteoclast depletion impairs neural crest proliferation and craniofacial morphogenesis"],"prefix":"10.1242","volume":"153","author":[{"given":"Felix","family":"Ma","sequence":"first","affiliation":[{"name":"The University of British Columbia 1 Craniofacial Science Graduate Program, Faculty of Dentistry , , Vancouver, BC V6T 1Z3 ,","place":["Canada"]},{"name":"Life Sciences Institute, The University of British Columbia 2 , Vancouver, BC V6T 1Z3 ,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rose Ru Jing","family":"Zhou","sequence":"additional","affiliation":[{"name":"Life Sciences Institute, The University of British Columbia 2 , Vancouver, BC V6T 1Z3 ,","place":["Canada"]},{"name":"The University of British Columbia 3 Doctor of Dental Medicine Program, Faculty of Dentistry , , Vancouver, BC V6T 1Z3 ,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Matthew","family":"Rosin","sequence":"additional","affiliation":[{"name":"Life Sciences Institute, The University of British Columbia 2 , Vancouver, BC V6T 1Z3 ,","place":["Canada"]},{"name":"The University of British Columbia 4 Department of Oral Biological and Medical Sciences, Faculty of Dentistry , , Vancouver, BC V6T 1Z3 ,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Iris","family":"Zhou","sequence":"additional","affiliation":[{"name":"Life Sciences Institute, The University of British Columbia 2 , Vancouver, BC V6T 1Z3 ,","place":["Canada"]},{"name":"The University of British Columbia 3 Doctor of Dental Medicine Program, Faculty of Dentistry , , Vancouver, BC V6T 1Z3 ,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sabrina","family":"Ownsworth","sequence":"additional","affiliation":[{"name":"Life Sciences Institute, The University of British Columbia 2 , Vancouver, BC V6T 1Z3 ,","place":["Canada"]},{"name":"The University of British Columbia 3 Doctor of Dental Medicine Program, Faculty of Dentistry , , Vancouver, BC V6T 1Z3 ,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rouzbeh Ostadsharif","family":"Memar","sequence":"additional","affiliation":[{"name":"The University of British Columbia 1 Craniofacial Science Graduate Program, Faculty of Dentistry , , Vancouver, BC V6T 1Z3 ,","place":["Canada"]},{"name":"Life Sciences Institute, The University of British Columbia 2 , Vancouver, BC V6T 1Z3 ,","place":["Canada"]},{"name":"The University of British Columbia 5 Division of Periodontics and Dental Hygiene, Faculty of Dentistry , , Vancouver, BC V6T 1Z3 ,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vincent B.","family":"Wong","sequence":"additional","affiliation":[{"name":"Life Sciences Institute, The University of British Columbia 2 , Vancouver, BC V6T 1Z3 ,","place":["Canada"]},{"name":"McGill University 6 Neuroscience Program, Faculty of Science , , Montreal, QC H3A 1A1 ,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0001-2650-4077","authenticated-orcid":false,"given":"Jessica M.","family":"Rosin","sequence":"additional","affiliation":[{"name":"Life Sciences Institute, The University of British Columbia 2 , Vancouver, BC V6T 1Z3 ,","place":["Canada"]},{"name":"The University of British Columbia 4 Department of Oral Biological and Medical Sciences, Faculty of Dentistry , , Vancouver, BC V6T 1Z3 ,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2026,5,6]]},"reference":[{"key":"2026050610240157200_DEV205423C1","doi-asserted-by":"publisher","first-page":"1517","DOI":"10.1002\/jbmr.354","article-title":"Osteal macrophages promote in vivo intramembranous bone healing in a mouse tibial injury model","volume":"26","author":"Alexander","year":"2011","journal-title":"J. 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