Septic arthritis of the temporomandibular joint (SATMJ) is a rare entity that has only been reported a few dozen times worldwide. However, SATMJ is an acute infectious disease and associated with a high risk for misdiagnosis, which results in severe complications for patients. The present study reported a case of an 83-year-old female patient with severe pyogenic orofacial infections caused by right-side SATMJ, which was not cured by abscess incision drainage and anti-microbial treatment. Finally, the clinical signs were resolved by condylectomy and debridement of the erosive septic TMJ, after which the function of the TMJ was completely restored. In conclusion, a timely and correct diagnosis with early treatment is critical to preventing acute complications and late sequelae of SATMJ. In addition, literature associated with this topic was reviewed and discussed.