The odontogenic keratocyst (OKC) is a developmental odontogenic cyst typically occurring in the jaws. Since the first description of OKC was published in 1956, the lesion has been of particular interest because of its specific histopathologic features, high recurrence rate, and aggressive behavior. Recurrences most commonly arise within bone at the site of the original cyst. However, as lining cells may find their way into surrounding tissues either from implantation during surgery or from cortical perforation recurrences may arise at a distance from the original cyst. Here, we report a rare case of recurrent OKC which was first developed in mandible and recurred within the masticatory space.