Shoulder pigmented villonodular synovitis (PVNS) is a severe clinical condition, while few studies have focused on this situation due to its rarity. This study was to investigate the efficacy of arthroscopic treatment of patients diagnosed with shoulder PVNS.From Jan 1st, 2010 to Dec. 31st, 2019, 6 patients (5 females and 1 male) diagnosed with shoulder PVNS underwent arthroscopic synovectomy in our hospital and combined rotator cuff repair was performed in 3 of them. The outcomes of this study include Constant score, Visual Analogue Scale (VAS), University of California, Los Angeles (UCLA) score and American Shoulder and Elbow Surgeons (ASES) score. The data were retrieved from the patients' medical records.With a mean follow-up of 52.0 months (range, 28-92 months), the mean difference of Constant, VAS, UCLA and ASES scores were 27.83 ± 21.60, 2.83 ± 2.56, 11.67 ± 10.93 and 17.83 ± 25.35, respectively. Statistically significant improvements were detected in all the patient-reported outcomes except ASES score. One of the patients suffered from recurrence. Two patients suffered from mild complications after the surgeries while both of them achieved satisfactory recovery finally.Arthroscopic synovectomy in the setting of shoulder PVNS can improve patients' function. A concurrent rotator cuff repair is recommended if it is needed. The conclusion still needs testifying by further high-quality research with larger sample size.© 2022. The Author(s).