We describe an unusual case of an odontogenic keratocyst (OKC) associated with an ungrafted left-sided alveolar cleft in a 10-year-old male patient. There is no previous report in the literature of OKC or other dental cysts associated with an alveolar cleft. We discuss the management of the OKC prior to secondary bone grafting and present this case to highlight the difficulty in the management of OKC concurrent with grafting of the alveolar cleft site, the proximity of unerupted permanent teeth, and possible treatment modalities.
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