The authors report a very rare patient with ectopic odontogenic keratocyst (OKC) in the temporal region that is distant form the mandible. Based on the interesting report, they discuss about the possible origin and illustrate the development of the ectopic OKC. It shows that the OKC could distally relapse with the help of temporal muscle. The surgeon should be more proactive to deal with the peripheral muscle of lesion.
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