This ruled out ligand activation as the mechanism underlying meningioma formation, and we notably observed a splice acceptor mutation (ENST00000593685.1:c.955-1G>C) in DYRK1B. DYRK1B was previously established as a negative regulator of Hh signaling, with a loss of this gene resulting in increases in expression of Hh pathway target genes and effectors30,31. Here, DYRK1B is linked to meningioma.