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Keratocystic odontogenic tumors (KCOTs) of the jaw affect more than 65% of patients with basal cell nevus syndrome (BCNS). Surgery frequently causes facial disfigurement and is not always curative. Most BCNS-related and some sporadic KCOTs have malignant activation of the Hedgehog signaling pathway.

The Use of Vismodegib to Shrink Keratocystic Odontogenic Tumors in Patients With Basal Cell Nevus Syndrome