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Vismodegib is the first-in-class inhibitor of the sonic hedgehog pathway useful in the treatment of locally advanced or metastatic basal cell carcinoma (BCC) that is not amenable to surgery and radiation therapy. Common adverse events of vismodegib, probably mechanism related, include alopecia (58%) as a reversible side effect. We report 2 cases of patients receiving vismodegib for the treatment of locally advanced BCCs that developed alopecia during treatment and describe clinical, dermoscopic, and reflectance confocal microscopy (RCM)