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We previously identified the novel ECM protein SNED1 as a promoter of breast cancer metastasis and showed that its level of expression negatively correlated with breast cancer patient survival. Here, we sought to identify the roles of SNED1 during murine development.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      We generated two novel\n                      <jats:italic>Sned1<\/jats:italic>\n                      knockout mouse strains and showed that\n                      <jats:italic>Sned1<\/jats:italic>\n                      is essential since homozygous ablation of the gene led to early neonatal lethality. Phenotypic analysis of the surviving knockout mice revealed a role for SNED1 in the development of craniofacial and skeletal structures since\n                      <jats:italic>Sned1<\/jats:italic>\n                      knockout resulted in growth defects, nasal cavity occlusion, and craniofacial malformations.\n                      <jats:italic>Sned1<\/jats:italic>\n                      is widely expressed in embryos, notably by cell populations undergoing epithelial\u2010to\u2010mesenchymal transition, such as the neural crest cells. We further show that mice with a neural\u2010crest\u2010cell\u2010specific deletion of\n                      <jats:italic>Sned1<\/jats:italic>\n                      survive, but display facial anomalies partly phenocopying the global knockout mice.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>\n                      Our results demonstrate requisite roles for SNED1 during development and neonatal survival. Importantly, the deletion of 2q37.3 in humans, a region that includes the\n                      <jats:italic>SNED1<\/jats:italic>\n                      locus, has been associated with facial dysmorphism and short stature.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/dvdy.258","type":"journal-article","created":{"date-parts":[[2020,10,4]],"date-time":"2020-10-04T06:55:27Z","timestamp":1601794527000},"page":"274-294","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":27,"title":["Knockout of the gene encoding the extracellular matrix protein\n                    <scp>SNED1<\/scp>\n                    results in early neonatal lethality and craniofacial malformations"],"prefix":"10.1002","volume":"250","author":[{"given":"Anna","family":"Barqu\u00e9","sequence":"first","affiliation":[{"name":"Department of Physiology and Biophysics College of Medicine, University of Illinois at Chicago  Chicago 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