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TranscriptomicsExpression profiling in WT vs CDKL5 mutated IPSCs
Mutations in the human CDKL5 gene have been associated with early onset seizure variant of Rett Syndrome. In order to investigate the potential involvement of CDKL5 in the regulation of gene expression, we compared expression profiles in WT vs CDKL5-mutated IPS clones from two patients (one male and...
ORGANISM(S): Homo sapiens 
2014-07-02 | E-MTAB-2223 | biostudies-arrayexpress
ProteomicsLF2 phosphorylation of the CDKL5 activation loop controls CDKL5’s activity
Cdkl5 Deficiency Disorder (CDD) is caused by variants in the protein kinase CDKL5, leading to symptoms such as seizures, developmental delay, and severe intellectual disability. The Chlamydomonas homologue of human CDKL5 is the flagellar protein LF5, whose absence results in a long flagella phenotyp...
ORGANISM(S): Chlamydomonas reinhardtii 
2025-11-15 | PXD068782 | Pride
Cilia Cdkl5 Phospho-proteome Flagella Cdd Chlamydomonas Cdk20 Lf5 Activation loop Lf2 Phosphorylation Kinase Proteome Cdkl5 deficiency disorder 
TranscriptomicsTranscription profiling by array of human clonal primary fibroblasts from patients with CDKL5 mutations
Mutations in the human CDKL5 gene have been shown to cause infantile spasms, as well as Rett syndrome-like phenotype. Because CDKL5 is subjected to X chromosome inactivation (XCI), individual cells from CDKL5 mutation girls either express the wild-type or mutant allele, likely resulting in different...
ORGANISM(S): Homo sapiens 
2011-07-23 | E-MEXP-2833 | biostudies-arrayexpress
ProteomicsLF2 phosphorylation of the CDKL5 activation loop controls CDKL5’s activity
Cdkl5 Deficiency Disorder (CDD) is caused by variants in the protein kinase CDKL5, leading to symptoms such as seizures, developmental delay, and severe intellectual disability. The Chlamydomonas homologue of human CDKL5 is the flagellar protein LF5, whose absence results in a long flagella phenotyp...
ORGANISM(S): Chlamydomonas reinhardtii 
2025-11-15 | PXD066796 | Pride
Cilia Cdkl5 Phospho-proteome Flagella Cdd Chlamydomonas Cdk20 Lf5 Activation loop Lf2 Phosphorylation Kinase Proteome Cdkl5 deficiency disorder 
ProteomicsQuantitative phosphoproteomic for CDKL5 substrates
Global phosphoproteomic screen to identify the first cellular substrates of CDKL5. CDKL5 knock-out U2OS cells and CDKL5 wt U2OS cells were generated for the TMT-based phosphoproteomic. Thi leads to the identification and further validation of several phosphopetides of MAP1S, CEP131 and CDKL5 itself....
ORGANISM(S): Homo sapiens (Human) 
2018-09-24 | PXD009374 | Pride
Cdkl5 substrates Phosphoproteomic Tmt Ms2 
ProteomicsChemical genetic identification of CDKL5 substrates reveals its role in neuronal microtubule dynamics
Loss-of-function mutations in CDKL5 kinase causes severe neurodevelopmental delay and early-onset seizures. Identification of CDKL5 substrates is key to understanding its function. Using chemical genetics, we found that CDKL5 phosphorylates three microtubule-associated proteins: MAP1S, EB2 and ARHGE...
ORGANISM(S): Mus musculus (Mouse) 
2018-09-18 | PXD010511 | Pride
Chemical genetics/ cdkl5/ map1s/ eb2/ microtubule dynamics 
ProteomicsLF2 phosphorylation of the CDKL5 activation loop controls CDKL5’s activity
Cdkl5 Deficiency Disorder (CDD) is caused by variants in the protein kinase CDKL5, leading to symptoms such as seizures, developmental delay, and severe intellectual disability. The Chlamydomonas homologue of human CDKL5 is the flagellar protein LF5, whose absence results in a long flagella phenotyp...
ORGANISM(S): Chlamydomonas reinhardtii 
2025-11-18 | PXD066877 | Pride
Chlamydomonas Cilia Cdkl5 deficiency disorder Ciliopathy Intraflagellar transport Kinase Flagella 
ProteomicsEpilepsy-linked kinase CDKL5 phosphorylates voltage-gated calcium channel Cav2.3, altering inactivation kinetics and neuronal excitability
Developmental and epileptic encephalopathies (DEEs) are a group of rare childhood disorders characterized by severe epilepsy and cognitive deficits. Numerous DEE genes have been discovered thanks to advances in genomic diagnosis, yet putative molecular links between these disorders are unknown. CDKL...
ORGANISM(S): Mus musculus (Mouse) 
2023-12-13 | PXD038505 | Pride
Cdkl5 Epilepsy Kinase Phosphoproteomics 
GenomicsBase editing restores CDKL5 expression and rescues neuronal deficits in a patient-derived model of CDKL5 deficiency disorder
Base editing restores CDKL5 expression and rescues neuronal deficits in a patient-derived model of CDKL5 deficiency disorder
PRJNA1438745 | ENA
ProteomicsCDKL5 substrates - Phosphoproteomic screening identifies physiological substrates of the CDKL5 kinase
Identification of CDKL5 substrates. Immuno-precipitated proteins were TMT labelled and analysed via LC-MS/MS.
ORGANISM(S): Homo sapiens (Human) 
2018-10-02 | PXD009327 | Pride
Cdkl5 Tmt Dlg5 map1s Cep131 
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