title: Unique spatiotemporal requirements for intraflagellar transport genes during forebrain development.
abstract: Primary cilia are organelles extended from virtually all cells and are required for the proper regulation of a number of canonical developmental pathways. The role in cortical development of proteins important for ciliary form and function is a relatively understudied area. Here we have taken a genetic approach to define the role in forebrain development of three intraflagellar transport proteins known to be important for primary cilia function. We have genetically ablated Kif3a, Ift88, and Ttc21b in a series of specific spatiotemporal domains. The resulting phenotypes allow us to draw several conclusions. First, we conclude that the Ttc21b cortical phenotype is not due to the activity of Ttc21b within the brain itself. Secondly, some of the most striking phenotypes are from ablations in the neural crest cells and the adjacent surface ectoderm indicating that cilia transduce critical tissue-tissue interactions in the developing embryonic head. Finally, we note striking differences in phenotypes from ablations only one embryonic day apart, indicating very discrete spatiotemporal requirements for these three genes in cortical development.
authors.affiliation: Division of Human Genetics, Department of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, United States of America.
authors.forename: John
authors.identifier: http://orcid.org/0000-0003-4457-480X
authors.initials: J
authors.lastname: Snedeker
authors.affiliation: Division of Developmental Biology, Department of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, United States of America.|Division of Plastic Surgery, Department of Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, United States of America.
authors.forename: Elizabeth N
authors.identifier: http://orcid.org/0000-0002-3970-799X
authors.initials: EN
authors.lastname: Schock
authors.affiliation: Division of Developmental Biology, Department of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, United States of America.|Division of Plastic Surgery, Department of Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, United States of America.
authors.forename: Jamie N
authors.identifier: http://orcid.org/0000-0002-2915-1588
authors.initials: JN
authors.lastname: Struve
authors.affiliation: Division of Developmental Biology, Department of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, United States of America.|Division of Plastic Surgery, Department of Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, United States of America.
authors.forename: Ching-Fang
authors.initials: CF
authors.lastname: Chang
authors.affiliation: Division of Human Genetics, Department of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, United States of America.
authors.forename: Megan
authors.initials: M
authors.lastname: Cionni
authors.affiliation: Department of Anatomy and Cell Biology, University of Kansas Medical Center, Kansas City, Kansas, United States of America.
authors.forename: Pamela V
authors.identifier: http://orcid.org/0000-0003-4619-4376
authors.initials: PV
authors.lastname: Tran
authors.affiliation: Division of Developmental Biology, Department of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, United States of America.|Division of Plastic Surgery, Department of Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, United States of America.
authors.forename: Samantha A
authors.initials: SA
authors.lastname: Brugmann
authors.affiliation: Division of Human Genetics, Department of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, United States of America.|Division of Developmental Biology, Department of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, United States of America.
authors.forename: Rolf W
authors.identifier: http://orcid.org/0000-0003-4512-6806
authors.initials: RW
authors.lastname: Stottmann
mesh_terms: D048868:Adaptor Proteins, Signal Transducing / Q000235:genetics*; D000818:Animals; D001692:Biological Transport / Q000235:genetics*; D002923:Cilia / Q000502:physiology*; D016547:Kinesins / Q000235:genetics*; D051379:Mice; D018345:Mice, Knockout; D016548:Prosencephalon / Q000196:embryology*; D025521:Tumor Suppressor Proteins / Q000235:genetics*
chemical_list: D048868:Adaptor Proteins, Signal Transducing; C490202:Kif3a protein, mouse; C490898:Tg737Rpw protein, mouse; C528451:Ttc21b protein, mouse; D025521:Tumor Suppressor Proteins; D016547:Kinesins