Thirty years ago, osteoarticular disorders which are related to various skin lesions such as pustulosis, psoriasis and acne have been reported in Japan and northwestern Europe. This disease entity is composed of three major disorders, which include the hyperostosis of the anterior chest wall, chronic multifocal recurrent osteomyelitis involving the metaphysis of long bone and pustulotic arthro-osteitis. In 1987, Kahn and Chamot first coined the term SAPHO syndrome (Synovitis, Acne, Pustulosis, Hyperostosis, Osteitis) . Early lesions of the SAPHO syndrome contain acute inflammation, edema, and prominent periosteal bone formation, and which is indistinguishable from ordinary bacterial osteomyelitis. In addition, late lesions of that demonstrate bony sclerosis. Therefore, it is necessary to avoid misdiagnosis as Paget's disease and Ewing sarcoma. We experienced a case of SAPHO syndrome and report the clinical, radiological and pathological findings in detail.