Mutations in human CILK1 (ciliogenesis-associated kinase 1) have been linked to both ciliopathies and epilepsy (Limerick et al., 2024) which is consistent with the disease manifestation resulting from mutations in ADGRV1. As in the case of pathogenic ADGRV1 mutations the ciliation rate, the ciliary length, and Shh signaling are altered by CILK1 mutations. Here, CILK1 is linked to ciliopathy.