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A tissue-specific role for intraflagellar transport genes during craniofacial development.

ABSTRACT: Primary cilia are nearly ubiquitous, cellular projections that function to transduce molecular signals during development. Loss of functional primary cilia has a particularly profound effect on the developing craniofacial complex, causing several anomalies including craniosynostosis, micrognathia, midfacial dysplasia, cleft lip/palate and oral/dental defects. Development of the craniofacial complex is an intricate process that requires interactions between several different tissues including neural crest cells, neuroectoderm and surface ectoderm. To understand the tissue-specific requirements for primary cilia during craniofacial development we conditionally deleted three separate intraflagellar transport genes, Kif3a, Ift88 and Ttc21b with three distinct drivers, Wnt1-Cre, Crect and AP2-Cre which drive recombination in neural crest, surface ectoderm alone, and neural crest, surface ectoderm and neuroectoderm, respectively. We found that tissue-specific conditional loss of ciliary genes with different functions produces profoundly different facial phenotypes. Furthermore, analysis of basic cellular behaviors in these mutants suggests that loss of primary cilia in a distinct tissue has unique effects on development of adjacent tissues. Together, these data suggest specific spatiotemporal roles for intraflagellar transport genes and the primary cilium during craniofacial development.

SUBMITTER: Schock EN

PROVIDER: S-EPMC5367710 | biostudies-literature | 2017

REPOSITORIES: biostudies-literature

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A tissue-specific role for intraflagellar transport genes during craniofacial development.

Schock Elizabeth N EN Struve Jaime N JN Chang Ching-Fang CF Williams Trevor J TJ Snedeker John J Attia Aria C AC Stottmann Rolf W RW Brugmann Samantha A SA

PloS one 20170327 3

Primary cilia are nearly ubiquitous, cellular projections that function to transduce molecular signals during development. Loss of functional primary cilia has a particularly profound effect on the developing craniofacial complex, causing several anomalies including craniosynostosis, micrognathia, midfacial dysplasia, cleft lip/palate and oral/dental defects. Development of the craniofacial complex is an intricate process that requires interactions between several different tissues including neu ...[more]

PMID: 28346501

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Dataset Information

A tissue-specific role for intraflagellar transport genes during craniofacial development.

Publications

A tissue-specific role for intraflagellar transport genes during craniofacial development.

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