An odontogenic keratocyst (OKC) was first described by Philipsen in 1956. They are benign cysts of odontogenic origin that behave aggressively and have a high recurrence rate.The present case report describes an unusual presentation of OKC as a multi-locular lesion in the anterior mandible.A 14-year-old male patient was referred to the oral maxillofacial surgery clinic in Tabuk Specialist Dental Centre by his orthodontist to evaluate a radiolucent lesion that had been identified in his lower anterior teeth during an OPG examination. The patient was medically fit and had multiple previous dental restorations. An intraoral examination revealed a small bony expansion in the cystic lesion on the buccal side. The panoramic radiograph showed well-defined multi-locular radiolucencies in the lower left canine area, despite there being no tooth resorption; however, there was a slight divergence noted between the teeth. An excisional biopsy was performed, and the subsequent histopathological examination revealed a cystic lesion diagnosed as an odontogenic keratocyst. The six-month follow-up OPG showed that the site had completely healed without any lesions recurring.OKCs can present at any age, irrespective of gender. The differential diagnosis included a lateral periodontal cyst or a radicular cyst when the tooth was not vital. In this case, the six-month follow-up OPG following surgery revealed no recurrence, although a close follow-up is recommended because of the high recurrence rate.