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Although odontogenic keratocysts are benign they are often locally destructive and tend to recur after conservative surgical treatment. They must therefore be distinguished from other cysts of the jaw. Keratocysts possess outpouchings and microscopic daughter cysts from which recurrences may arise. Histologic examination is essential for diagnosis since the appearances on roentgenograms and at operation usually do not reveal the true nature of the lesion. Since many nondental surgeons and pathologists are unaware of odontogenic keratocysts a case is presented in which surgical treatment was originally conservative and finally relatively radical.