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Environmental or genetic interferences can impair neurulation, resulting in clinically significant birth defects known collectively as neural tube defects. The\n                      <jats:italic>Fuz<\/jats:italic>\n                      gene encodes a subunit of the CPLANE complex, a macromolecular planar polarity effector required for ciliogenesis. Ablation of\n                      <jats:italic>Fuz<\/jats:italic>\n                      in mouse embryos results in exencephaly and spina bifida, including dysmorphic craniofacial structures due to defective cilia formation and impaired Sonic Hedgehog signaling.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      We demonstrate that knocking\n                      <jats:italic>Fuz<\/jats:italic>\n                      out during embryonic mouse development results in a hypoplastic hindbrain phenotype, displaying abnormal rhombomeres with reduced length and width. This phenotype is associated with persistent reduction of ventral neuroepithelial stiffness in a notochord adjacent area at the level of the rhombomere 5. The formation of cranial and paravertebral ganglia is also impaired in these embryos.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>\n                      This study reveals that hypoplastic hindbrain development, identified by abnormal rhombomere morphology and persistent loss of ventral neuroepithelial stiffness, precedes exencephaly in\n                      <jats:italic>Fuz<\/jats:italic>\n                      ablated murine mutants, indicating that the gene\n                      <jats:italic>Fuz<\/jats:italic>\n                      has a critical function sustaining normal neural tube development and neuronal differentiation.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/dvdy.702","type":"journal-article","created":{"date-parts":[[2024,3,19]],"date-time":"2024-03-19T08:19:55Z","timestamp":1710836395000},"page":"846-858","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Disruption of\n                    <scp>\n                      <i>Fuz<\/i>\n                    <\/scp>\n                    in mouse embryos generates hypoplastic hindbrain development and reduced cranial nerve ganglia"],"prefix":"10.1002","volume":"253","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-4982-5631","authenticated-orcid":false,"given":"Carlo Donato","family":"Caiaffa","sequence":"first","affiliation":[{"name":"Center for Precision Environmental Health Baylor College of Medicine  Houston Texas USA"},{"name":"Department of Pediatrics, Dell Pediatric Research Institute University of Texas at Austin Dell Medical School  Austin Texas 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