OBJECTIVE To describe an unusual location for a common benign salivary gland neoplasm. METHODS Case report and literature review. METHODS We describe an unusual lesion found in the infratemporal fossa on computed tomography of the facial skeleton, including the clinical presentation, workup, and surgical approach. A complete PubMed search of atypical presentations of pleomorphic adenoma was performed. The literature was also reviewed for all neoplasms involving the infratemporal fossa, pterygomaxillary fissure, and pterygopalatine fossa. RESULTS Pleomorphic adenoma has been reported to arise in such diverse locations as the nasal septum, main stem bronchus, trachea, lacrimal gland and external auditory canal. One case has been reported to occur in pterygopalatine fossa. No cases of pleomorphic adenoma involving the infratemporal fossa have been reported. There are, however, a number of other neoplasms that can arise, or more frequently, directly invade, this region. Malignancies can often have an insidious presentation that is initially referred to other disease entities. CONCLUSIONS Though rare, there are a variety of neoplasms that can involve the infratemporal fossa. Patients with malignancies of this region usually present late in their disease course with invasion into surrounding structures. Diagnosis and approach to this region is difficult considering its concealed location.