Abstract
OBJECTIVE: The health effects of SAPHO (synovitis, acne, pustulosis, hyperostosis, and osteitis) syndrome and chronic nonbacterial osteomyelitis (CNO) have not been well studied. We assessed health-related quality of life (HRQOL) in adults with SAPHO-CNO and performed a review of EQ-5D questionnaire outcomes among similar chronic rheumatic and inflammatory diseases. METHODS: We enrolled patients in the first US-based SAPHO-CNO prospective registry and assessed their HRQOL using the EQ-5D index and EQ-5D visual analog scale (VAS). A focused scoping review was performed of EQ-5D-related outcomes among disease cohorts with phenotypic similarities to SAPHO-CNO. RESULTS: In the 138 participants with SAPHO-CNO, mean age was 37.7 (SD 15.0) years with a mean disease duration of 6.4 (SD 6.2) years. All subjects had musculoskeletal involvement (with 43% having spine involvement) and 46% had skin involvement. Mean 5-level EQ-5D index value was 0.75 (SD 0.16) and mean EQ-VAS was 63.0 (SD 21.0). Most patients were affected by pain/discomfort (86%), difficulty in carrying out usual activities (65%), and anxiety/depression (54%); over one-third reported problems in all 5 EQ-5D domains. These adults with SAPHO-CNO had similarly reduced EQ-5D and VAS values, and similar high proportion of perceived problems, as in other inflammatory disease cohorts. CONCLUSION: The HRQOL among adults with SAPHO-CNO is low, with more than half of patients experiencing greater impact on perceived pain, difficulty performing usual activities, and feelings of anxiety or depression. Future therapies should address the multidimensional nature of SAPHO-CNO, targeting HRQOL as a major outcome.