Treatment options for single-system Langerhans cell histiocytosis (LCH) range from observation to chemotherapy. Among these options, corticosteroid injection is appealing due to the feasibility, preservation of structure and function as well as the low complication rate. Therefore, this study aimed to investigate the effectiveness of intralesional injection of triamcinolone acetonide (TA) for the treatment of craniofacial LCH and present a typical case. 11 patients diagnosed with LCH of the jaws who received intralesional injection of TA at Hospital of Stomatology, Sun Yat-sen University from April 2014 to May 2021 were involved. Clinicopathologic data were collected and analyzed. The 11 subjects included 7 males and 4 females (mean age 14.6±16.9 y). Most lesions were located in the mandible (n=9, 81.8%). The effective rate of intralesional injection of TA was 90.9%. The average initial dosage and average accumulative dosage were 33.2±21.0 and 99.5±88.7 mg, respectively. The mean time needed for complete ossification of the lesion was 12.5±8.2 months. The mean follow-up time was 20±13.4 months. Although the effective rate was similar between children and adults (100% versus 75%, P=0.364), the initial dosage, accumulative dosage, number of injections, and time needed for complete ossification of the lesion were all lower in children (P<0.05). In conclusion, intralesional injection of TA is a suitable treatment option for craniofacial LCH, especially in children. Studies that incorporate a larger patient cohort should be conducted to verify the current findings.