Cyclin-Dependent Kinase-Like-5 (CDKL5) Deficiency Disorder (CDD) is a severe X-linked neurodegenerative disease characterized primarily by seizures, which begin in the first months of life, associated with low muscle tone, severe motor rigidity deficient language acquisition, and progressive intellectual disability. A zebrafish cdkl5 mutant line (cdkl5sa21938) has been recently characterized and shown to reproduce defects in behavior, morphological and molecular characteristics similar to those observed in CDD. Accordingly, we propose to exploit this model to identify pharmacological drugs suitable to treat/relief symptoms of CDKL5 deficiency.