Unique spatiotemporal requirements for intraflagellar transport genes during forebrain development

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 gene...

Full description

Saved in:
Bibliographic Details
Published in:PloS one 2017-03, Vol.12 (3), p.e0173258-e0173258
Main Authors: Snedeker, John, Schock, Elizabeth N, Struve, Jamie N, Chang, Ching-Fang, Cionni, Megan, Tran, Pamela V, Brugmann, Samantha A, Stottmann, Rolf W
Format: Article
Language:English
Subjects:
Ablation
Adaptor Proteins, Signal Transducing - genetics
Animals
Arthritis
Binding sites
Biological Transport - genetics
Biology and Life Sciences
Brain
Cell cycle
Cell development (Biology)
Cilia
Cilia - physiology
Cortex
Defects
Developmental biology
Ectoderm
Embryos
Forebrain
Genes
Genetics
Hospitals
Kidney diseases
Kinesin - genetics
Medicine and Health Sciences
Mice
Mice, Knockout
Microtubules
Mutation
Neural crest
Organelles
Pediatrics
Physiological aspects
Prosencephalon - embryology
Proteins
Stem cells
Transport
Tumor Suppressor Proteins - genetics
Citations: Items that this one cites
Items that cite this one
Online Access:Get full text
Tags: Add Tag
No Tags, Be the first to tag this record!
Description
Summary: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.
ISSN:1932-6203
1932-6203
DOI:10.1371/journal.pone.0173258