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GOSHS is due to recessive mutations in <jats:italic>KIAA1279<\/jats:italic>, which encodes kinesin family member 1 binding protein (KIF1BP, also known as KBP). We examined the effects of inactivation of <jats:italic>Kif1bp<\/jats:italic> in mice. Mice lacking Kif1bp died shortly after birth, and exhibited smaller brains, olfactory bulbs and anterior commissures, and defects in the vagal and sympathetic innervation of the gut. <jats:italic>Kif1bp<\/jats:italic> was found to interact with <jats:italic>Ret<\/jats:italic> to regulate the development of the vagal innervation of the stomach. Although newborn <jats:italic>Kif1bp<\/jats:italic><jats:sup>\u2212\/\u2212<\/jats:sup> mice had neurons along the entire bowel, the colonization of the gut by neural crest-derived cells was delayed. The data show an essential <jats:italic>in vivo<\/jats:italic> role for KIF1BP in axon extension from some neurons, and the reduced size of the olfactory bulb also suggests additional roles for KIF1BP. Our mouse model provides a valuable resource to understand GOSHS.<\/jats:p>","DOI":"10.1038\/s41598-017-16965-3","type":"journal-article","created":{"date-parts":[[2017,11,24]],"date-time":"2017-11-24T14:57:51Z","timestamp":1511535471000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":17,"title":["Kif1bp loss in mice leads to defects in the peripheral and central nervous system and perinatal death"],"prefix":"10.1038","volume":"7","author":[{"given":"Caroline S.","family":"Hirst","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8925-7894","authenticated-orcid":false,"given":"Lincon A.","family":"Stamp","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Annette J.","family":"Bergner","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marlene M.","family":"Hao","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mai X.","family":"Tran","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jan M.","family":"Morgan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Matthias","family":"Dutschmann","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2183-5360","authenticated-orcid":false,"given":"Andrew M.","family":"Allen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"George","family":"Paxinos","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Teri M.","family":"Furlong","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sonja J.","family":"McKeown","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0457-8710","authenticated-orcid":false,"given":"Heather M.","family":"Young","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2017,11,30]]},"reference":[{"key":"16965_CR1","first-page":"485","volume":"36","author":"AS Brooks","year":"1999","unstructured":"Brooks, A. 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