Syphilitic arthritis is a very rare disease with diverse symptoms, therefore, prone to be of miss diagnosis or misdiagnosis. In this paper, we report a 61-year-old female patient with fever, right shoulder swelling and pain, and mobility limitation for 10 months. Ar- throscopy revealed synovial hyperplasia with some nodular and jelly-like changes, rotator cuff rupture, destruction of the humeral head and glenoid complicated with partial defects. The hyperplastic synovial tissue was debrided by shavers, and the broken cartilage and scar of the humeral head and glenoid were removed. Histopathological examination of the synovium revealed leptospira. Combined with this case, this paper also mentioned the clinical and imaging features, diagnosis, differential diagnosis, and treatment of syphilitic arthritis by a literature review to provide reference for clinicians.