While the fundamental role of biallelic NF2 loss in meningiomas from NF2 patients and approximately 50% of sporadic meningiomas has long been recognized [27, 28], a spectrum of recurrent oncogenic driver mutations including in SMO, KLF4, TRAF7, AKT1, or PIK3CA has more recently been identified, and are mutually exclusive with NF2 alterations. Here, SMO is linked to meningioma.