Odontogenic keratocysts make up 4%-12% of all odontogenic cysts. Most cysts are sporadic but sometimes they arise in the context of basal cell nevus syndrome (Gorlin syndrome). Most odontogenic keratocysts arise in the posterior region of the mandible, but they can occur anywhere in the jaw. In rare instances, they are located peripherally in the gingiva. Even more rare, they are found in the soft tissues of the mouth. There have been a few case reports and small case series of such peripheral odontogenic keratocysts. Some controversy exists as to whether these truly represent a peripheral counterpart of the intraosseous odontogenic keratocysts and if their origin is at all odontogenic. We hereby present two cases of peripheral odontogenic keratocysts, both being located in the soft tissue of the buccal mucosa, and review the literature on peripheral odontogenic keratocysts.