Bone infection, particularly of the skull and the long bones of the legs and arms, despite being a common characteristic of tertiary stage syphilis and congenital syphilis in the past, is seldom encountered clinically due to effective antibiotic therapy. We report a case of a 62-year-old man who presented with one-month acute pain in the left leg. Treponema pallidum particle agglutination test was positive, and radiography showed a pathological fracture of the left tibiofibula. Surgical debridement and biopsy of the bone marrow were performed and a pathological diagnosis of syphilis osteomyelitis was entertained. The patient's symptoms resolved completely after a six-week course of penicillin. We identified 17 previously published cases of bone lesions of syphilis, eight of which had syphilitic osteomyelitis, seven were syphilitic osteitis and two had syphilitic periostitis. We suggest that bone lesions such as osteomyelitis caused by syphilis can be the only manifestation of late latent syphilis.