顎関節強直症を呈したSAPHO症候群の1例

• Published in: 日本口腔外科学会雑誌 Vol. 68; no. 9; pp. 370 - 377
• Main Authors: 新國, 農, 児玉, 泰光, 大貫, 尚志, 髙木, 律男, 上野山, 敦士, 西山, 秀昌
• Format: Journal Article
• Language: Japanese
• Published: 公益社団法人 日本口腔外科学会 20.09.2022
• Subjects: SAPHO症候群; びまん性硬化性骨髄炎; 顎関節強直症; 顎関節授動術
• ISSN: 0021-5163; 2186-1579
• DOI: 10.5794/jjoms.68.370

SAPHO syndrome is a disorder characterized by aseptic inflammatory bone, joint and skin lesions. Bone lesions in the maxillofacial region often occur in the mandible as diffuse sclerosing osteomyelitis (DSO). It is rarely associated with temporomandibular joint (TMJ) ankylosis. We report an exceptionally rare case of SAPHO syndrome with TMJ ankylosis treated with arthroplasty. The patient was a 50-year-old man with a history of SAPHO syndrome. He was referred to our department in April 2009 due to pain in the left mandible with trismus. His mouth opening was 9 mm. The patient was diagnosed with left mandibular osteomyelitis and started treatment with NSAIDs, but he soon stopped visiting our hospital. He visited the hospital again in November 2013. His mouth opening was 5 mm. Computed tomography showed that the left mandibular condyle had bony adhesions to the glenoid fossa and we diagnosed TMJ ankylosis. Since he also had sternoclavicular joint pain, treatment for SAPHO syndrome was performed in our hospital’s department of collagen disease. The pain soon disappeared, but the mouth opening decreased further to 2 mm, so arthroplasty was performed in May 2017. The mouth opening was 30 mm after the operation. Two years passed after the operation, and the mouth opening was 22 mm. Careful observation is necessary for SAPHO syndrome with DSO, taking the development of TMJ ankylosis into consideration. In severe cases, it is necessary to consider surgical treatment.