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Updated: Jan 19, 2026
Identification of Cyclin-dependent Kinase 1 Specific Phosphorylation Sites by an In Vitro Kinase Assay
Published on: May 3, 2018
Ciliogenesis associated kinase 1: targets and functions in various organ systems
Zheng Fu1, Casey D Gailey1, Eric J Wang1
1Department of Pharmacology, University of Virginia, Charlottesville, VA, USA.
Abstract:
Ciliogenesis associated kinase 1 (CILK1) was previously known as intestinal cell kinase because it was cloned from that origin. However, CILK1 is now recognized as a widely expressed and highly conserved serine/threonine protein kinase. Mutations in the human CILK1 gene have been associated with ciliopathies, a group of human genetic disorders with defects in the primary cilium. In mice, both Cilk1 knock-out and Cilk1 knock-in mutations have recapitulated human ciliopathies. Thus, CILK1 has a fundamental role in the function of the cilium. Several candidate substrates have been proposed for CILK1 and the challenge is to relate these to the mutant phenotypes. In this review, we summarize what is known about CILK1 functions and targets, and discuss gaps in current knowledge that motivate further experimentation to fully understand the role of CILK1 in organ development in humans.
Insights
Ciliogenesis associated kinase 1 (CILK1) is crucial for primary cilium function. Mutations in CILK1 cause human ciliopathies, highlighting its role in organ development.
Area of Science:
- Biochemistry
- Molecular Biology
- Genetics
Background:
- Ciliogenesis associated kinase 1 (CILK1), formerly intestinal cell kinase, is a conserved serine/threonine protein kinase.
- CILK1 is widely expressed and essential for primary cilium function.
Purpose of the Study:
- To review CILK1 functions and targets.
- To discuss knowledge gaps regarding CILK1's role in human organ development and ciliopathies.
Main Methods:
- Review of existing literature on CILK1.
- Analysis of genetic studies involving CILK1 mutations in humans and mice.
Main Results:
- Human CILK1 gene mutations are linked to ciliopathies.
- Cilk1 knockout and knock-in mouse models replicate human ciliopathies, confirming CILK1's fundamental role.
Conclusions:
- CILK1 plays a critical role in primary cilium function and organ development.
- Further research is needed to connect CILK1 substrates to observed mutant phenotypes and fully elucidate its function.
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