Objectives: To report a giant cell tumor (GCT) of temporal bone and infratemporal fossa and to review the literature pertinent to the care of such patients. Study Design: Case report and review of the literature. Methods: A review of the literature was conducted using Pubmed and the key words temporal bone, GCT, infratemporal fossa, and recurrent GCT. Medical records from an index case were analyzed and presented in the context of the available literature. Results: Six case reports over 23 years illustrate that these benign locally destructive lesions may originate in the temporal bone with extension into the infratemporal fossa. More commonly, GCTs involve the distal ends of long bones. We present a 30 year-old male who developed a temporal bone GCT with infratemporal fossa extension 12 years after undergoing successful surgical treatment of a GCT of the femur. Our index case differs from prior reported cases by surgical approach and by the occurrence of an infratemporal fossa GCT as well as a previous GCT at a separate anatomical location. Complete removal was achieved but required resection of the zygomatic arch and dissection of all upper facial nerve branches. This was tolerated well with acceptable functional and cosmetic results. The patient is disease free after 24 months without facial nerve deficits. Conclusion: Due to the risk for recurrence, complete resection of GCTs of the temporal bone and infratemporal fossa is advocated. Surgical techniques that allow for visualization of the facial nerve and increase surgical access can enhance overall clinical success with limited post-operative morbidity.