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Background: Chronic pain is a common clinical feature that accompanies osteopathy. Knowledge and attitudes of both patients and their treating clinicians would influence the patients' outcomes and pain control. Objective: This systematic review will provide an understanding of the knowledge and attitudes of patients and clinicians towards musculoskeletal pain accompanying osteopathy. Methods: Different keywords were used to search the medical literature, including: “knowledge” OR “Attitude” AND “Osteopathy” AND “Pain” AND “patient” OR “clinician." The search databases included Medline, Embase PubMed, and SCOPUS. The following step was reviewing the appearing results to ensure that they were original research articles that examined the knowledge and attitudes about chronic musculoskeletal pain with osteopathy. All the eligible studies should mention the type of participants examined (either patients or clinicians). Results: A total of 89 studies were obtained. After removing review articles and choosing original research studies solely, 11 studies appeared from the filtration process. Eight research articles were eligible. All the included studies had a quantitative cross-sectional design. Only health care professionals were asked about osteopathy, where all the studies included osteopaths from different countries, except one study that included physiotherapists. Osteopaths knew about the benefits of osteopathy, particularly for lower back pain; however, their knowledge about biopsychosocial factors requires improvement. Conclusion: Knowledge about osteopathy benefits for controlling chronic musculoskeletal pain should be improved even among osteopaths. Awareness campaigns are also needed for patients in orthopedic and physiotherapy clinics about osteopathy.