A pediatric case of diffuse sclerosing osteomyelitis of the mandible treated successfully by anti-TNF-α therapy

Abstract

SHIMOIDE Takeshi ・ ENOMOTO Akifumi ・ MATSUNAGA Kazuhide IWASAKI Sanae ・ OKAMOTO Reiko ・ HAMADA Suguru : Diffuse sclerosing osteomyelitis of the mandible (DSO) is a non-bacterial osteomyelitis that has been considered one of the symptoms of chronic recurrent multifocal osteomyelitis or synovitis, acne, pustulosis, hyperostosis and osteitis syndrome, which are autoinflammatory diseases. DSO is clinically difficult to diagnose, and no clear consensus has been established regarding its management. We herein present a pediatric case of DSO successfully treated by anti-TNF-α therapy. A 13-year-old Japanese child was referred to our clinic because of swelling of the right mandibular gingiva. Antibiotics were administered as an initial treatment, but were ineffective. We used hyperbaric oxygen therapy and performed mandibular corticotomy; however, these treatments were also ineffective. Incisional biopsy of the mandible region was performed, revealing DSO. Treatment with a corticosteroid was begun, and clinical symptoms dramatically decreased. However, we could not decrease the corticosteroid dose owing to the recurrence of inflammatory symptoms. Bisphosphonate or disease-modifying antirheumatic drug treatment was also effective but was stopped owing to adverse reactions. Then, the patient was treated with anti-TNF-α antibody. After administration, remarkable improvement of inflammatory findings was confirmed. In addition, no major side effect or complication was encountered, and a complete response has been achieved for 2 years. This case provides a basis for the future appropriate management of DSO using anti-TNF-α antibody in pediatric patients. : DSO (慢性び漫性硬化性下顎骨骨髄炎) SAPHO syndrome (SAPHO 症候群) CRMO (慢性再発性多発性骨 髄炎) autoinflammatory disease (自己炎症性疾患) anti-TNF-α antibody (抗 TNF-α 抗体) 近畿大学医学部附属病院歯科口腔外科 (主任：濱田 傑教授