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J Clin Immunol ; 41(3): 565-575, 2021 04.
Article in English | MEDLINE | ID: mdl-33392854

ABSTRACT

SAPHO (synovitis, acne, pustulosis, hyperostosis, and osteitis) syndrome shows a wide variability in musculoskeletal and cutaneous manifestations, and it is therefore underrecognized and misdiagnosed in the clinic due to a lack of specific markers. In this study, we aimed to identify specific biomarkers by screening serum autoantibodies in SAPHO patients with a 17K human whole-proteome microarray. The serum anti-Sp17 autoantibody was identified and verified to be a specific biomarker in patients with SAPHO syndrome. Indeed, the level of the anti-Sp17 autoantibody was significantly increased in patients with active SAPHO compared to patients with an inactive disease and healthy controls (P < 0.05). Additionally, serum anti-Sp17 autoantibody levels correlated with those of serum hypersensitive C-reactive protein (hsCRP), the erythrocyte sedimentation rate (ESR), and ß-crosslaps (ß-CTx) in patients with active SAPHO disease. Moreover, anti-Sp17 autoantibody levels were markedly decreased after anti-inflammatory treatment with pamidronate disodium, which downregulated levels of hsCRP and ESR in patients with active SAPHO. Thus, serum levels of the anti-Sp17 autoantibody might serve as a specific biomarker for the diagnosis of SAPHO syndrome or for monitoring the disease status.


Subject(s)
Acquired Hyperostosis Syndrome/blood , Acquired Hyperostosis Syndrome/diagnosis , Autoantibodies/blood , Autoantibodies/immunology , Biomarkers , Calmodulin-Binding Proteins/immunology , Membrane Proteins/immunology , Acquired Hyperostosis Syndrome/drug therapy , Acquired Hyperostosis Syndrome/etiology , Adult , Bone Density Conservation Agents/therapeutic use , Bone and Bones/metabolism , Calmodulin-Binding Proteins/genetics , Case-Control Studies , Female , Humans , Inflammation Mediators/metabolism , Male , Membrane Proteins/genetics , Middle Aged , Pamidronate/therapeutic use , Prognosis , Proteome , Proteomics/methods , Proteomics/standards , Sensitivity and Specificity
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