The effects of treatment with FRAX486 on CDD‐related alterations were assessed in vitro (100 nM for 48 h) on primary hippocampal cultures from Cdkl5‐knockout male mice (Cdkl5‐KO) and in vivo (20 mg/Kg, s.c. for 5 days) on Cdkl5‐KO heterozygous females (Cdkl5‐Het). This evidence concerns the gene CDKL5 and craniodiaphyseal dysplasia.