ArticlePediatric nephrology (Berlin, Germany)2008
Defects in ciliary localization of Nek8 is associated with cystogenesis.
Article in Pediatric nephrology (Berlin, Germany), 2008. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 26 papers.
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Who cites it
26 citing papers in PubMed, 39 citations in OpenAlex.
- NEK8, a NIMA-family protein kinase at the core of the ciliary INV complex.Cell communication and signaling : CCS · 2025Review
- TNF-alpha promotes cilia elongation via mixed lineage kinases signaling in mouse fibroblasts and human RPE-1 cells.Cytoskeleton (Hoboken, N.J.) · 2024Article
- An unexpected role for the conserved ADAM-family metalloprotease ADM-2 in Caenorhabditis elegans molting.PLoS genetics · 2022Article
- Nephronophthisis-Pathobiology and Molecular Pathogenesis of a Rare Kidney Genetic Disease.Genes · 2021Review
- Ciliary Genes in Renal Cystic Diseases.Cells · 2020Review
- Novel fibrillar structure in the inversin compartment of primary cilia revealed by 3D single-molecule superresolution microscopy.Molecular biology of the cell · 2020Article
- Actin organization and endocytic trafficking are controlled by a network linking NIMA-related kinases to the CDC-42-SID-3/ACK1 pathway.PLoS genetics · 2018Article
- Mechanisms for nonmitotic activation of Aurora-A at cilia.Biochemical Society transactions · 2017Review
- Article
- Effects of TORC1 Inhibition during the Early and Established Phases of Polycystic Kidney Disease.PloS one · 2016Article
- C. elegans NIMA-related kinases NEKL-2 and NEKL-3 are required for the completion of molting.Developmental biology · 2015Article
- The tumor suppressor pVHL down-regulates never-in-mitosis A-related kinase 8 via hypoxia-inducible factors to maintain cilia in human renal cancer cells.The Journal of biological chemistry · 2015Article
- Abnormalities associated with progressive aortic vascular dysfunction in chronic kidney disease.Frontiers in physiology · 2015Article
- Ciliopathies: the trafficking connection.Traffic (Copenhagen, Denmark) · 2014Review
- Compartments within a compartment: what C. elegans can tell us about ciliary subdomain composition, biogenesis, function, and disease.Organogenesis · 2014Review
- Wnt and planar cell polarity signaling in cystic renal disease.Organogenesis · 2014Review
- Loss of the ciliary kinase Nek8 causes left-right asymmetry defects.Journal of the American Society of Nephrology : JASN · 2013Article
- A novel mutation causing nephronophthisis in the Lewis polycystic kidney rat localises to a conserved RCC1 domain in Nek8.BMC genomics · 2012Article
- The Nek8 protein kinase, mutated in the human cystic kidney disease nephronophthisis, is both activated and degraded during ciliogenesis.Human molecular genetics · 2012Article
- Stages of ciliogenesis and regulation of ciliary length.Differentiation; research in biological diversity · 2012Review
Corrections and comments
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Authors and funding
6 authors at 5 institutions in 2 countries.
Funding
Abstract
Mutations in the human NIMA (Never in Mitosis gene A)-related kinase 8 (Nek8) are associated with a rare form of the juvenile renal cystic disease, nephronophthisis type 9, and mutations in murine Nek8 cause renal cysts in jck mice. Cystogenesis involves dysfunctional ciliary signaling, and we have previously reported that Nek8 localizes to the primary cilium in mouse kidney epithelial cells. We now report that in developing mouse kidney, Nek8 is detected in the cilia of a subset of ureteric-bud-derived tubules at embryonic day (E)15.5. An increasing proportion of ureteric-bud-derived tubules express ciliary Nek8 until E18.5. Postnatal day 1 and 7 Nek8 is observed with equal frequency in both ureteric-bud and non-ureteric-bud-derived tubules. To investigate the cell biological consequences of kinase-deficient and jck mutant forms of Nek8, we transiently expressed green fluorescent protein (GFP)-tagged constructs in vitro. Mutations in the kinase and C-terminal domains of Nek8 adversely affected ciliary targeting but did not affect ciliogenesis or ciliary length. Consistent with these in vitro observations, kidneys from homozygous jck mice revealed reduced ciliary expression of Nek8 compared with kidneys from heterozygous (unaffected) mice. These data indicate that the ciliary localization of Nek8 in a subset of ureteric-bud-derived kidney tubules is essential for maintaining the integrity of those tubules in the mammalian kidney.
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