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About the Authors:
Ashley Snouffer
Contributed equally to this work with: Ashley Snouffer, Desmond Brown, Hankyu Lee
Roles Formal analysis, Investigation, Methodology, Writing - original draft
Affiliation: Department of Genetics, University of Georgia, Athens, GA, United States of America
ORCID http://orcid.org/0000-0003-2179-8288
Desmond Brown
Contributed equally to this work with: Ashley Snouffer, Desmond Brown, Hankyu Lee
Roles Investigation, Methodology
Current address: Department of Neurosurgery, Mayo School of Graduate Medical Education, Rochester, MN, United States of America
Affiliation: Department of Molecular Biology, Princeton University, Princeton, NJ, United States of America
ORCID http://orcid.org/0000-0002-2896-1722
Hankyu Lee
Contributed equally to this work with: Ashley Snouffer, Desmond Brown, Hankyu Lee
Roles Formal analysis, Investigation
Affiliation: College of Pharmacy, Dongguk University-Seoul, Goyang, Korea
Jonathon Walsh
Roles Investigation, Methodology
Affiliation: Department of Genetics, University of Georgia, Athens, GA, United States of America
ORCID http://orcid.org/0000-0002-4277-7657
Floria Lupu
Roles Formal analysis, Investigation, Writing - review & editing
Affiliation: Department of Genetics, University of Georgia, Athens, GA, United States of America
ORCID http://orcid.org/0000-0003-1222-9884
Ryan Norman
Roles Investigation
Affiliation: Department of Molecular Biology, Princeton University, Princeton, NJ, United States of America
Karl Lechtreck
Roles Conceptualization, Methodology
Affiliation: Department of Cellular Biology, University of Georgia, Athens, GA, United States of America
Hyuk Wan Ko
Roles Conceptualization, Formal analysis, Funding acquisition, Investigation, Supervision, Writing - original draft, Writing - review & editing
* E-mail: [email protected] (HWK); [email protected] (JE)
Affiliation: College of Pharmacy, Dongguk University-Seoul, Goyang, Korea
ORCID http://orcid.org/0000-0003-1753-1900
Jonathan Eggenschwiler
Roles Conceptualization, Formal analysis, Funding acquisition, Investigation, Methodology, Project administration, Writing - original draft, Writing - review & editing
* E-mail: [email protected] (HWK); [email protected] (JE)
Affiliation: Department of Genetics, University of Georgia, Athens, GA, United States of America
ORCID http://orcid.org/0000-0001-6444-8865Abstract
The Hedgehog (Hh) signaling pathway plays a key role in cell fate specification, proliferation, and survival during mammalian development. Cells require a small organelle, the primary cilium, to respond properly to Hh signals and the key regulators of Hh signal transduction exhibit dynamic localization to this organelle when the pathway is activated. Here, we investigate the role of Cell Cycle Related kinase (CCRK) in regulation of cilium-dependent Hh signaling in the mouse. Mice mutant for Ccrk exhibit a variety of developmental defects indicative of inappropriate regulation of this pathway. Cell biological, biochemical and genetic analyses indicate that CCRK is...