Glandular odontogenic cyst (GOC) associated with ameloblastoma is an exceedingly rare histologic presentation with no known clinical significance or treatment applications. Four cases have been reported, three in the mandible and one in the maxilla. The age range is 14-65 and with male predilection. All four presented with swellings and two with pain. We add one more case to the literature of a 58-year old male presenting with an expansile multilocular radiolucency between teeth #19-23. The ameloblastomatous changes in this case are consistent with those of a unicystic ameloblastoma-mural subtype. Although the histologic changes are those of a unicystic ameloblastoma, the clinical and radiographic findings are not. This case therefore presents a clinical challenge with regards to treatment planning for recurrence and prognosis. We conclude that treatment for GOC therefore be based on individual clinical presentation.