A case is presented of a 14-year-old boy with aneurysmal bone cyst of the tuberculum articulare of the temporomandibular joint (TMJ). This disease rarely involves the skull, and involvement of temporal bone is even more rare. To our knowledge, only 22 cases have been reported in the literature. This is the first case of aneurysmal bone cyst of the tuberculum articulare of the TMJ described in the literature. Keywords: aneurysmal bone cyst, temporal bone, temporomandibular joint, tuberculum articulare