An extensive Odontogenic Keratocyst in the left maxilla: a case report

Odontogenic Keratocysts (OKC) are benign cystic lesions that are known to be locally aggressive and have a high recurrence rate. Here we describe a case of a particularly extensive OKC.

A 28 year-old female presented to the Oral and Maxillofacial department with a 2-month history of facial swelling and pain. On examination, there was a left mid-facial swelling which was extremely tender on palpation, with intra-oral buccal expansion. Medically, she had depression and was a non-smoker. An OPG and CT neck with contrast was carried out; this revealed a substantial lesion, filling the left maxillary sinus, in association with a superiorly impacted UL8 tooth located just below the orbital apex. The lesion had cystic characteristics, measuring 6.6cm vertically, and 5.8 x 3.8cm in a transverse dimension. The lesion was shown to project medially into the nasal cavity, resulting in septal deviation to the right. Superiorly, there was associated convex bulging of the orbital floor.

Subsequently, antibiotics were prescribed; enucleation of the cyst and extraction of UL8 was carried out. The pathology result was in favour of Odontogenic Keratocyst. Infection reoccurred 1 month later which led to a washout of the cystic cavity, placement of a drain, extraction of a resorbed UL6 and additional antibiotic therapy. Gabapentin was then commenced due to associated neuropathic pain. 3 months following initial surgery, further enucleation of a 1x1cm residual cyst was carried out. 6 months later, a repeat CT scan revealed mildly progressive bony infill in line with ongoing healing and no evidence of recurrence.

This case highlights the aggressive nature of the OKC despite their slow growth. The presence of such an expansile lesion in the maxillary sinus with an associated ectopic tooth just below the orbital apex is rare. Close follow-up to detect recurrence is important.