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References from Activation of the ciliary kinase CDKL5 is mediated by the cyclin-dependent kinase CDK20/LF2 to control flagellar length. Local targets link to admitted publications; unresolved targets remain external evidence.
Prevalence and onset of comorbidities in the CDKL5 disorder differ from Rett syndrome
10.1186/s13023-016-0418-y · 2016 · External reference
CDKL5 regulates flagellar length and localizes to the base of the flagella in Chlamydomonas
10.1091/mbc.e12-10-0718 · 2013 · External reference
Ciliopathies
10.1101/cshperspect.a028191 · 2017 · External reference
Cilia and diseases
10.1093/biosci/biu174 · 2014 · External reference
Cilia structure and function in human disease
10.1016/j.coemr.2024.100509 · 2024 · External reference
Primary ciliary dyskinesia
10.1016/j.lpm.2023.104171 · 2023 · External reference
The mouse ortholog of EFHC1 implicated in juvenile myoclonic epilepsy is an axonemal protein widely conserved among organisms with motile cilia and flagella
10.1016/j.febslet.2004.12.070 · 2005 · External reference
Roles of primary cilia in the developing brain
10.3389/fncel.2019.00218 · 2019 · External reference
CDKL family kinases have evolved distinct structural features and ciliary function
10.1016/j.celrep.2017.12.083 · 2018 · External reference
Phenotypic characterization of Cdkl5-knockdown neurons establishes elongated cilia as a functional assay for CDKL5 Deficiency Disorder
10.1016/j.neures.2021.10.001 · 2022 · External reference
Flow blockage disrupts cilia-driven fluid transport in the epileptic brain
10.1007/s00401-022-02463-y · 2022 · External reference
Proteomic analysis of a eukaryotic cilium
10.1083/jcb.200504008 · 2005 · External reference
The centriolar satellite protein AZI1 interacts with BBS4 and regulates ciliary trafficking of the BBSome
10.1371/journal.pgen.1004083 · 2014 · External reference
Acute versus chronic loss of mammalian Azi1/Cep131 results in distinct ciliary phenotypes
2013 · External reference
Chemical genetic identification of CDKL5 substrates reveals its role in neuronal microtubule dynamics
10.15252/embj.201899763 · 2018 · External reference
Phosphoproteomic screening identifies physiological substrates of the CDKL5 kinase
10.15252/embj.201899559 · 2018 · External reference
CDKL5/Stk9 kinase inactivation is associated with neuronal developmental disorders
10.1093/hmg/ddi391 · 2005 · External reference
CDKL5 belongs to the same molecular pathway of MeCP2 and it is responsible for the early-onset seizure variant of Rett syndrome
10.1093/hmg/ddi198 · 2005 · External reference
Cyclin-Dependent Kinase-Like 5 (CDKL5): possible cellular signalling targets and involvement in CDKL5 deficiency disorder
10.1155/2020/6970190 · 2020 · External reference
A CDK-related kinase regulates the length and assembly of flagella in Chlamydomonas
10.1083/jcb.200610022 · 2007 · External reference
The DYF-5 RCK and CDKL-1 CDKL5 kinases contribute differentially to shape distinct sensory neuron cilia morphologies in C. elegans
2022 · External reference
CDKL kinase regulates the length of the ciliary proximal segment
10.1016/j.cub.2021.03.068 · 2021 · External reference
De novo variants in CDKL1 and CDKL2 are associated with neurodevelopmental symptoms
10.1016/j.ajhg.2025.02.019 · 2025 · External reference
A phylogenetic analysis of the CDKL protein family unravels its evolutionary history and supports the Drosophila model of CDKL5 deficiency disorder
10.3389/fcell.2025.1582684 · 2025 · External reference
CDKL5 deficiency disorder in males: five new variants and review of the literature
10.1016/j.ejpn.2021.04.007 · 2021 · External reference
CDKL5-related disorders: from clinical description to molecular genetics
10.1159/000331333 · 2012 · External reference
Developmental changes in ciliary composition during gametogenesis in Chlamydomonas
10.1091/mbc.e22-02-0033 · 2022 · External reference
Cilia disassembly with two distinct phases of regulation
10.1016/j.celrep.2015.02.044 · 2015 · External reference
FLS2 is a CDK-like kinase that directly binds IFT70 and is required for proper ciliary disassembly in Chlamydomonas
10.1371/journal.pgen.1008561 · 2020 · External reference
Protein kinases: evolution of dynamic regulatory proteins
10.1016/j.tibs.2010.09.006 · 2011 · External reference
Loss of CDKL5 disrupts kinome profile and event-related potentials leading to autistic-like phenotypes in mice
10.1073/pnas.1216988110 · 2012 · External reference
A novel MAP kinase regulates flagellar length in Chlamydomonas
10.1016/s0960-9822(03)00415-9 · 2003 · External reference
Flagellar length control.
10.1016/b978-0-12-370873-1.00042-3 · 2009 · External reference
Analysis of protein kinases by Phos-tag SDS-PAGE
10.1016/j.jprot.2022.104485 · 2022 · External reference
Advances in Phos-tag-based methodologies for separation and detection of the phosphoproteome
10.1016/j.bbapap.2014.10.004 · 2015 · External reference
Less label, more free: approaches in label-free quantitative mass spectrometry
10.1002/pmic.201000553 · 2011 · External reference
Cell Cycle-Related Kinase (CCRK) regulates ciliogenesis and Hedgehog signaling in mice
10.1371/journal.pgen.1006912 · 2017 · External reference
Protein arginine methyltransferases interact with intraflagellar transport particles and change location during flagellar growth and resorption
10.1091/mbc.e16-11-0774 · 2017 · External reference
A protein methylation pathway in Chlamydomonas flagella is active during flagellar resorption
10.1091/mbc.e08-05-0470 · 2008 · External reference
CDKL5 kinase controls transcription-coupled responses to DNA damage
10.15252/embj.2021108271 · 2021 · External reference
Molecular basis of tubulin transport within the cilium by IFT74 and IFT81
10.1126/science.1240985 · 2013 · External reference
Intraflagellar transport proteins 172, 80, 57, 54, 38, and 20 form a stable tubulin-binding IFT-B2 complex
10.15252/embj.201593164 · 2016 · External reference
Getting tubulin to the tip of the cilium: one IFT train, many different tubulin cargo-binding sites?
10.1002/bies.201400007 · 2014 · External reference
Assembly of FAP93 at the proximal axoneme in Chlamydomonas cilia
10.1002/cm.21818 · 2024 · External reference
SADB phosphorylation of gamma-tubulin regulates centrosome duplication
10.1038/ncb1921 · 2009 · External reference
SnRK2 protein kinases—key regulators of plant response to abiotic stresses
10.1089/omi.2011.0091 · 2011 · External reference
The methylome of motile cilia
2024 · External reference
Ultrastructural differences impact cilia shape and external exposure across cell classes in the visual cortex
10.1016/j.cub.2024.04.043 · 2024 · External reference
Systematic proteomic analysis of LRRK2-mediated Rab GTPase phosphorylation establishes a connection to ciliogenesis
10.7554/elife.31012 · 2017 · External reference
Association of common variants in the Joubert syndrome gene (AHI1) with autism
10.1093/hmg/ddn291 · 2008 · External reference
Trisomy 21 represses cilia formation and function
10.1016/j.devcel.2018.07.008 · 2018 · External reference
Disruption of sonic hedgehog signaling in Ellis-van Creveld dwarfism confers protection against bipolar affective disorder
10.1038/mp.2014.118 · 2015 · External reference
Functional variants of CFAP410 affect the DNA damage response leading to motor neuron degeneration–implications for ALS
10.1016/j.isci.2025.113338 · 2025 · External reference
Mutations in NEK1 cause ciliary dysfunction as a novel pathogenic mechanism in amyotrophic lateral sclerosis
10.1186/s13024-025-00848-7 · 2025 · External reference
C21ORF2 mutations point towards primary cilia dysfunction in amyotrophic lateral sclerosis
10.1093/brain/awae331 · 2025 · External reference
Cell motility: deaf Drosophila keep the beat
10.1016/j.cub.2003.09.047 · 2003 · External reference
Abnormal length of cilia as a possible cause of defective mucociliary clearance
1985 · External reference
CCRK/CDK20 regulates ciliary retrograde protein trafficking via interacting with BROMI/TBC1D32
10.1371/journal.pone.0258497 · 2021 · External reference
Activity-dependent phosphorylation of CDKL5 at serine 407 regulates synaptogenesis and plasticity
10.1007/s12264-023-01066-1 · 2023 · External reference
Subcellular distribution of cyclin-dependent kinase-like 5 (CDKL5) is regulated through phosphorylation by dual specificity tyrosine-phosphorylation-regulated kinase 1A (DYRK1A)
10.1016/j.bbrc.2016.11.048 · 2017 · External reference
Regulation of flagellar assembly and length in Chlamydomonas by LF4, a MAPK-related kinase
10.1096/fj.201802375rr · 2019 · External reference
DYF-5/MAK-dependent phosphorylation promotes ciliary tubulin unloading
10.1073/pnas.2207134119 · 2022 · External reference
Centrosomal protein CEP104 (Chlamydomonas FAP256) moves to the ciliary tip during ciliary assembly
2013 · External reference
Regulation of cilium length and intraflagellar transport by the RCK-kinases ICK and MOK in renal epithelial cells
10.1371/journal.pone.0108470 · 2014 · External reference
ICK is essential for cell type-specific ciliogenesis and the regulation of ciliary transport
10.1002/embj.201488175 · 2014 · External reference
Negative regulation of ciliary length by ciliary male germ cell-associated kinase (Mak) is required for retinal photoreceptor survival
10.1073/pnas.1009437108 · 2010 · External reference
Cytochrome f and plastocyanin: their sequence in the photosynthetic electron transport chain of Chlamydomonas reinhardi
10.1073/pnas.54.6.1665 · 1965 · External reference
Nutritional studies with Chlamydomonas reinhardi
10.1111/j.1749-6632.1953.tb30261.x · 1953 · External reference
Isolation of Chlamydomonas flagella and flagellar axonemes
10.1016/0076-6879(86)34096-5 · 1986 · External reference
Direct in situ protein tagging in Chlamydomonas reinhardtii utilizing TIM, a method for CRISPR/Cas9-based targeted insertional mutagenesis
2022 · External reference
An engineered Streptomyces hygroscopicus aph 7″ gene mediates dominant resistance against hygromycin B in Chlamydomonas reinhardtii
10.1078/14344610260450136 · 2002 · External reference
TIM, a targeted insertional mutagenesis method utilizing CRISPR/Cas9 in Chlamydomonas reinhardtii
10.1371/journal.pone.0232594 · 2020 · External reference
Accurate structure prediction of biomolecular interactions with AlphaFold 3
10.1038/s41586-024-07487-w · 2024 · External reference
UCSF ChimeraX: structure visualization for researchers, educators, and developers
10.1002/pro.3943 · 2021 · External reference
UCSF ChimeraX: meeting modern challenges in visualization and analysis
10.1002/pro.3235 · 2018 · External reference
Fiji: an open-source platform for biological-image analysis
10.1038/nmeth.2019 · 2012 · External reference
Analysis of protein phosphorylation using Phos-Tag gels
10.1002/cpps.64 · 2018 · External reference
NIH Image to ImageJ: 25 years of image analysis
10.1038/nmeth.2089 · 2012 · External reference
Ependymal cell differentiation, from monociliated to multiciliated cells
10.1016/bs.mcb.2015.01.004 · 2015 · External reference
A polymerase chain reaction based method for detecting Mycoplasma/Acholeplasma contaminants in cell culture
10.1016/s0167-7012(99)00107-4 · 2000 · External reference
T5 exonuclease-dependent assembly offers a low-cost method for efficient cloning and site-directed mutagenesis
2019 · External reference
Sustained expression of α1-antitrypsin after transplantation of manipulated hematopoietic stem cells
10.1165/rcmb.2007-0133oc · 2008 · External reference
Optimized sgRNA design to maximize activity and minimize off-target effects of CRISPR-Cas9
10.1038/nbt.3437 · 2016 · External reference
CHOPCHOP v3: expanding the CRISPR web toolbox beyond genome editing
10.1093/nar/gkz365 · 2019 · External reference
Improved vectors and genome-wide libraries for CRISPR screening
10.1038/nmeth.3047 · 2014 · External reference
Mutational analysis of the phototransduction pathway of Chlamydomonas reinhardtii
10.1083/jcb.131.2.427 · 1995 · External reference
Portrait of a species: Chlamydomonas reinhardtii
10.1534/genetics.105.044503 · 2005 · External reference
Genetic analysis of long-flagella mutants of Chlamydomonas reinhardtii
10.1093/genetics/118.4.637 · 1988 · External reference
Protter: interactive protein feature visualization and integration with experimental proteomic data
10.1093/bioinformatics/btt607 · 2014 · External reference
