ABSTRACT: I will briefly give an overview how we use genetically modified mice and human induced pluripotent stem cells (iPSC)‐derived cells/brain organoids to model neurological diseases, such as developmental brain disorders as well as frontotemporal dementia (FTD) and amyotrophic lateral sclerosis (ALS) neurodegenerative diseases. Then I will mainly talk about skull‐brain crosstalk via meningeal lymphatics focusing on Saethre‐Chotzen syndrome, a congenital craniofacial disorder with brain dysfunctions. We identified meningeal lymphatics as a pathological factor and a therapeutic target for neurocognitive defects in craniosynostosis, providing novel in‐sights into the skull‐brain communications.