Introduction: The botryoid odontogenic cyst (BOC) is a developmental cyst of odontogenic epithelial origin considered as a rare multilocular variety of lateral periodontal cyst (WHO 1992). This cystic lesion site is in the periodontal space of vital teeth.
Observation: A 21-year-old woman consulted for a swelling of the anterior maxillary region. The clinical examination revealed bucal swelling extending from the left central incisor to the first left premolar. The mucosa was normal. The adjacent teeth were vital. X-ray revealed a bilocular radiolucency extending between the roots of teeth 21 and 24. The lesion was enucleated, under local anaesthesia via a vestibular approach. Histology was typical of a BOC with cystic spaces lined by squamous epithelium with thickening and clear cells. Forty-eight months after surgery, there was sign of recurrence.
Discussion: BOC is known to be a recurrent odontogenic cyst. Several cases of multiple recurrences have been reported up to nine years after the initial surgery. Long-term follow-up is thus mandatory.