Kikuchi Tomohiro, Fujii Hiroyuki, Fujita Akifumi, Sugiyama Tomoko, Sugimoto Hideharu
Department of Radiology, Jichi Medical University, School of Medicine, 3311-1 Yakushiji, Shimotsuke, Tochigi 329-0498, Japan.
Department of Dentistry, Oral and Maxillofacial Surgery, Jichi Medical University, School of Medicine, 3311-1 Yakushiji, Shimotsuke, Tochigi 329-0498, Japan.
Case Rep Radiol. 2018 Jun 13;2018:9142362. doi: 10.1155/2018/9142362. eCollection 2018.
Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a disorder characterized by pustular skin lesions and osteoarticular lesions. Mandibular involvement of SAPHO syndrome is clinically rare, and it is difficult to reach a diagnosis of SAPHO syndrome from only mandibular manifestations. This report describes the case of a 26-year-old woman who presented with mandibular osteitis. Orthopantomogram and computed tomography showed sclerotic change of the right body of the mandible with periosteal reaction without odontogenic infection, which suggested the possibility of SAPHO syndrome. Detailed medical interview found that she had a history of palmoplantar pustulosis treated at a local dermatology clinic and additional bone scintigraphy showed diffuse increased uptake in the right mandible, as well as in the sternum and the sternocostoclavicular joints. She was eventually diagnosed as having SAPHO syndrome. We should consider SAPHO syndrome when we encounter a patient with mandibular osteitis of unknown etiology.
滑膜炎、痤疮、脓疱病、骨肥厚和骨炎(SAPHO)综合征是一种以脓疱性皮肤病变和骨关节病变为特征的疾病。SAPHO综合征累及下颌骨在临床上较为罕见,仅根据下颌骨表现很难确诊为SAPHO综合征。本报告描述了一名26岁女性下颌骨炎病例。曲面体层摄影和计算机断层扫描显示右下颌骨体部硬化改变伴骨膜反应,无牙源性感染,提示可能为SAPHO综合征。详细的病史询问发现她曾在当地皮肤科诊所治疗掌跖脓疱病,进一步的骨闪烁显像显示右下颌骨以及胸骨和胸锁关节弥漫性摄取增加。她最终被诊断为SAPHO综合征。当遇到病因不明的下颌骨炎患者时,我们应考虑SAPHO综合征。