Vismodegib is useful to treat locally advanced or metastatic basal cell carcinomas (BCCs),1 but to our knowledge, its use as a neoadjuvant to shrink BCCs before surgery has not been reported. This case illustrates the role of vismodegib as a neoadjuvant agent. In addition, this case highlights the fact that a patient with Gorlin syndrome can develop resistant BCCs while taking vismodegib, a phenomenon not widely recognized although recently reported.2