Unique spatiotemporal requirements for intraflagellar transport genes during forebrain development

John Snedeker, Elizabeth N. Schock, Jamie N. Struve, Ching Fang Chang, Megan Cionni, Pamela V. Tran, Samantha A. Brugmann, Rolf W. Stottmann

Research output: Contribution to journalArticlepeer-review

24 Scopus citations

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 tissueDtissue 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.This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

Original languageEnglish
Article numbere0173258
JournalPLoS ONE
Volume12
Issue number3
DOIs
StatePublished - Mar 2017

Bibliographical note

Publisher Copyright:
© 2017 Haniffa et al.

Funding

FundersFunder number
Institute of Neurological Disorders and Stroke National Advisory Neurological Disorders and Stroke CouncilT32NS007453
Institute of Neurological Disorders and Stroke National Advisory Neurological Disorders and Stroke Council

    ASJC Scopus subject areas

    • General

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