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It is associated with recessive mutations in the gene encoding kinesin family member 1-binding protein (KIF1BP, also known as KIFBP). The encoded protein regulates axon microtubules dynamics, kinesin attachment and mitochondrial biogenesis, but it is not clear how its loss could lead to microcephaly. We identified KIF1BP in the interactome of citron kinase (CITK, also known as CIT), a protein produced by the primary hereditary microcephaly 17 (MCPH17) gene. KIF1BP and CITK interact under physiological conditions in mitotic cells. Similar to CITK, KIF1BP is enriched at the midbody ring and is required for cytokinesis. The association between KIF1BP and CITK can be influenced by CITK activity, and the two proteins may antagonize each other for their midbody localization. KIF1BP knockdown decreases microtubule stability, increases KIF23 midbody levels and impairs midbody localization of KIF14, as well as of chromosome passenger complex. These data indicate that KIF1BP is a CITK interactor involved in midbody maturation and abscission, and suggest that cytokinesis failure may contribute to the microcephaly phenotype observed in GOSHS.<\/jats:p>","DOI":"10.1242\/jcs.250902","type":"journal-article","created":{"date-parts":[[2021,5,20]],"date-time":"2021-05-20T11:23:46Z","timestamp":1621509826000},"source":"Crossref","is-referenced-by-count":11,"title":["Goldberg\u2013Shprintzen syndrome protein KIF1BP is a CITK interactor implicated in cytokinesis"],"prefix":"10.1242","volume":"134","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-7735-3010","authenticated-orcid":false,"given":"Gianmarco","family":"Pallavicini","sequence":"first","affiliation":[{"name":"Neuroscience Institute Cavalieri Ottolenghi, Turin 10123, Italy"},{"name":"Department of Neuroscience \u2018Rita Levi Montalcini\u2019, University of Turin, Turin 10126, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marta","family":"Gai","sequence":"additional","affiliation":[{"name":"Department of Molecular Biotechnology and Health Sciences, University of Turin, Turin 10126, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2966-9675","authenticated-orcid":false,"given":"Giorgia","family":"Iegiani","sequence":"additional","affiliation":[{"name":"Neuroscience Institute Cavalieri Ottolenghi, Turin 10123, Italy"},{"name":"Department of Neuroscience \u2018Rita Levi Montalcini\u2019, University of Turin, Turin 10126, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gaia Elena","family":"Berto","sequence":"additional","affiliation":[{"name":"Neuroscience Institute Cavalieri Ottolenghi, Turin 10123, Italy"},{"name":"Department of Neuroscience \u2018Rita Levi Montalcini\u2019, University of Turin, Turin 10126, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Annie","family":"Adrait","sequence":"additional","affiliation":[{"name":"Univ. Grenoble Alpes, Commissariat \u00e0 l'\u00e9nergie atomique et aux \u00e9nergies alternatives (CEA), Institut national de la sant\u00e9 et de la recherche m\u00e9dicale (INSERM), Interdisciplinary Research Institute of Grenoble (IRIG), Laboratoire Biologie \u00e0 Grande Echelle (BGE), 38000 Grenoble, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yohann","family":"Cout\u00e9","sequence":"additional","affiliation":[{"name":"Univ. 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