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A retrospective study of bone scintigraphy in the follow-up of patients with synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome: is it useful to repeat bone scintigraphy for disease assessment?

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Abstract

Objectives

To investigate the value of repeated bone scintigraphy in the follow-up of patients with synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome and to characterize the changing pattern of osteoarticular lesions revealed by bone scintigraphy.

Method

Twenty-four patients with SAPHO syndrome who had repetitively undergone bone scintigraphy and tests of inflammatory markers (erythrocyte sedimentation rate (ESR) and/or C-reactive protein (CRP)) were included in this retrospective study. The change in accumulation number was recorded as the difference in the number of accumulation sites between consecutive bone scintigraphy. The visual analog scale (VAS) for pain and medications prescribed were also reviewed. The relationships of the change in accumulation number with medication prescribed and change in ESR or CRP were analyzed.

Results

Twenty-four and 23 patients had follow-up tests of ESR and CRP, from which 30 and 28 follow-up data were obtained, containing the corresponding changes in ESR and CRP, respectively. A decrease in total accumulation number observed by bone scintigraphy was rarely observed, while decreases in ESR, CRP, and VAS were predominant. The accumulation number had significantly increased over time (follow-up with ESR: r = 0.389, p = 0.034; follow-up with CRP: r = 0.438, p = 0.020), in accordance with an “imprinting” pattern, while the inflammatory markers and VAS for pain predominantly decreased. There was no significant association between the change in accumulation number (local/total) and the change in ESR or CRP values (p > 0.05) or medications used for SAPHO (p > 0.05).

Conclusions

This retrospective cohort study of 24 SAPHO patients demonstrated an “imprinting” pattern on bone scintigraphy, without a correlation to the decrease in inflammatory markers, patient disease assessment, or treatment type. Thus, repeated bone scintigraphy did not contribute an additional clinical value for the follow-up of patients with SAPHO.

Key Points

• In a cohort of 24 SAPHO patients, repeated bone scintigraphy revealed a continuous increase in tracer accumulation number, indicating an “imprinting” pattern.

• The change in tracer accumulation number, defined as the difference in the number of accumulation sites between consecutive bone scintigraphy measurements, was inconsistent with the change in ESR, CRP, or VAS for pain.

• The medications prescribed for SAPHO did not seem to contribute to a decrease in accumulation number.

• Repeated bone scintigraphy did not seem to be useful for the assessment of disease activity in patients with SAPHO.

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References

  1. Chamot AM, Benhamou CL, Kahn MF, Beraneck L, Kaplan G, Prost A (1987) Acne-pustulosis-hyperostosis-osteitis syndrome. Results of a national survey. 85 cases. Rev Rhum Mal Osteoartic 54:187–196

    CAS  PubMed  Google Scholar 

  2. Zimmermann P, Curtis N (2016) Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome - a challenging diagnosis not to be missed. J Inf Secur 72(Suppl):S106–S114

    Google Scholar 

  3. Cianci F, Zoli A, Gremese E, Ferraccioli G (2017) Clinical heterogeneity of SAPHO syndrome: challenging diagnose and treatment. Clin Rheumatol 36:2151–2158

    Article  Google Scholar 

  4. Depasquale R, Kumar N, Lalam RK, Tins BJ, Tyrrell PN, Singh J, Cassar-Pullicino VN (2012) SAPHO: what radiologists should know. Clin Radiol 67:195–206

    Article  CAS  Google Scholar 

  5. Zhang S, Li C, Zhang S, Li L, Zhang W, Dong Z, Zhang W (2019) Serum levels of proinflammatory, anti-inflammatory cytokines, and RANKL/OPG in synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome. Mod Rheumatol 29:523–530

    Article  CAS  Google Scholar 

  6. Schaub S, Sirkis HM, Kay J (2016) Imaging for synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome. Rheum Dis Clin N Am 42:695–710

    Article  Google Scholar 

  7. Van den Wyngaert T, Strobel K, Kampen WU, Kuwert T, van der Bruggen W, Mohan HK, Gnanasegaran G, Delgado-Bolton R, Weber WA, Beheshti M, Langsteger W, Giammarile F, Mottaghy FM, Paycha F (2016) The EANM practice guidelines for bone scintigraphy. Eur J Nucl Med Mol Imaging 43:1723–1738

    Article  Google Scholar 

  8. Nguyen MT, Borchers A, Selmi C, Naguwa SM, Cheema G, Gershwin ME (2012) The SAPHO syndrome. Semin Arthritis Rheum 42:254–265

    Article  Google Scholar 

  9. Cao Y, Li C, Yang Q, Wu N, Xu P, Li Y, Shi X, Pan Q, Wu X, Sun X, Zhao Y, Gu Z, Yuan S, Zuo Y, Li L, Zhang W, Hao W, Liu J, Jing H, and Zhang W (2019) Three patterns of osteoarticular involvement in SAPHO syndrome: a cluster analysis based on whole body bone scintigraphy of 157 patients. Rheumatology (Oxford) 58:1047–1055

  10. Rohekar G, Inman RD (2006) Conundrums in nosology: synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome and spondylarthritis. Arthritis Rheum 55:665–669

    Article  CAS  Google Scholar 

  11. Gheita TA, Azkalany GS, Kenawy SA, Kandeel AA (2015) Bone scintigraphy in axial seronegative spondyloarthritis patients: role in detection of subclinical peripheral arthritis and disease activity. Int J Rheum Dis 18:553–559

    Article  Google Scholar 

  12. Cao Y, Li C, Xu W, Wu X, Sun X, Zhang W, Jing H, Gu Z, Yuan S, Li L, Zuo Y, Liu J, Wu Z, Dong Z, Hao W, Zhang W, Wu N (2018) Spinal and sacroiliac involvement in SAPHO syndrome: a single center study of a cohort of 354 patients. Semin Arthritis Rheum 48:990–996

  13. Cohen J (1992) A power primer. Psychol Bull 112:155–159

    Article  CAS  Google Scholar 

  14. Li C, Zuo Y, Wu N, Li L, Li F, Zhang W, Xu W, Zhao X, Jing H, Pan Q, Zhou W, Shi X, Fan Y, Wang J, Liu S, Liu Z, Zhang F, Zeng X, Chen H, Zhang S, Liu J, Qiu G, Wu Z, Dong Z, Zhang W (2016) Synovitis, acne, pustulosis, hyperostosis and osteitis syndrome: a single centre study of a cohort of 164 patients. Rheumatology (Oxford) 55:1023–1030

    Article  CAS  Google Scholar 

  15. Colina M, Govoni M, Orzincolo C, Trotta F (2009) Clinical and radiologic evolution of synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome: a single center study of a cohort of 71 subjects. Arthritis Rheum 61:813–821

    Article  Google Scholar 

  16. Witt M, Meier J, Hammitzsch A, Proft F, Schulze-Koops H, Grunke M (2014) Disease burden, disease manifestations and current treatment regimen of the SAPHO syndrome in Germany: results from a nationwide patient survey. Semin Arthritis Rheum 43:745–750

    Article  Google Scholar 

  17. Firinu D, Garcia-Larsen V, Manconi PE, Del Giacco SR (2016) SAPHO syndrome: current developments and approaches to clinical treatment. Curr Rheumatol Rep 18:35

    Article  Google Scholar 

  18. Li C, Zhao Y, Zuo Y, Zhou Y, Zhang F, Liu S, Zhu Q, Chen J, Zhang W, Xu W, Gu Z, Li L, Li F, Tao W, Cao Y, Sun X, Jing H, Chen H, Zhang S, Dong Z, Liu J, Shi X, Hao W, Qiu G, Zhang W, Wu N, and Wu Z (2019) Efficacy of bisphosphonates in patients with synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome: a prospective open study. Clin Exp Rheumatol 37:663–669

  19. Leone A, Cassar-Pullicino VN, Casale R, Magarelli N, Semprini A, Colosimo C (2015) The SAPHO syndrome revisited with an emphasis on spinal manifestations. Skelet Radiol 44:9–24

    Article  Google Scholar 

  20. Yang DC, Ratani RS, Mittal PK, Chua RS, Pate SM (2002) Radionuclide three-phase whole-body bone imaging. Clin Nucl Med 27:419–426

    Article  Google Scholar 

  21. Klett R, Grau K, Puille M, Matter HP, Lange U, Steiner D, Bauer R (2000) Comparison of HIG scintigraphy and bloodpool scintigraphy using HDP in arthritic joint disease. Nuklearmedizin 39:33–37

    CAS  PubMed  Google Scholar 

  22. Ming Wei Tang SL, Chen L, James Cheng Chung W (2019) Treatment for Sapho syndrome: a multimodality therapy. Biomed J Sci Tech Res 14:BJSTR. MS.ID.002548

    Google Scholar 

  23. Baraliakos X, Braun J (2016) Imaging scoring methods in axial spondyloarthritis. Rheum Dis Clin N Am 42:663–678

    Article  Google Scholar 

  24. Sun X, Li C, Cao Y, Shi X, Li L, Zhang W, Wu X, Wu N, Jing H, Zhang W (2018) F-18 FDG PET/CT in 26 patients with SAPHO syndrome: a new vision of clinical and bone scintigraphy correlation. J Orthop Surg Res 13:120

    Article  Google Scholar 

  25. Pichler R, Weiglein K, Schmekal B, Sfetsos K, Maschek W (2003) Bone scintigraphy using Tc-99m DPD and F18-FDG in a patient with SAPHO syndrome. Scand J Rheumatol 32:58–60

    Article  Google Scholar 

  26. Anzola-Fuentes LK, Chianelli M, Galli F, Glaudemans AW, Martin Martin L, Todino V, Migliore A, Signore A (2016) Somatostatin receptor scintigraphy in patients with rheumatoid arthritis and secondary Sjogren’s syndrome treated with infliximab: a pilot study. EJNMMI Res 6:49

    Article  CAS  Google Scholar 

Download references

Funding

This work was supported by the National Natural Science Foundation of China (81822030, 81501852, 81472046, 81472045, 81772299), the Beijing Natural Science Foundation (7172175), the CAMS Initiative for Innovative Medicine (2017-I2M-3-001, 2017-I2M-2-001, 2016-I2M-3-003 and 2016-I2M-2-006), the National Key Research and Development Program of China (2016YFC0901501), and the Capital Medical Research and Development Fund (2016-4-40112).

Author information

Author notes

  1. Chen Li, Lun Wang and Nan Wu contributed equally to this work.

Authors and Affiliations

  1. Department of Traditional Chinese Medicine, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, No.1 Shuaifuyuan, Beijing, 100730, China

    Chen Li

  2. Institute of Clinical Medicine, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, No.9 Dongdansantiao, Beijing, 100006, China

    Lun Wang

  3. Department of Orthopedic Surgery, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, No.1 Shuaifuyuan, Beijing, 100730, China

    Nan Wu

  4. Medical Research Center of Orthopedics, Chinese Academy of Medical Sciences, No.1 Shuaifuyuan, Beijing, 100730, China

    Nan Wu

  5. Beijing Key Laboratory for Genetic Research of Skeletal Deformity, No.1 Shuaifuyuan, Beijing, 100730, China

    Nan Wu

  6. Department of Radiology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, No.1 Shuaifuyuan, Beijing, 100730, China

    Yihan Cao

  7. Department of Rheumatology and Clinical Immunology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, No.1 Shuaifuyuan, Beijing, 100730, China

    Xiaochuan Sun & Wen Zhang

  8. Department of Nuclear Medicine, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, No.1 Shuaifuyuan, Beijing, 100730, China

    Hongli Jing

Authors
  1. Chen Li
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Contributions

Study conception and design: Chen Li, Lun Wang, Nan Wu.

Acquisition of data: Lun Wang, Yihan Cao, Xiaochuan Sun, Hongli Jing.

Analysis and interpretation of data: Chen Li, Lun Wang, Nan Wu, Hongli Jing, Wen Zhang.

Drafting of manuscript: Lun Wang, Chen Li.

Critical revision: Nan Wu, Hongli Jing, Wen Zhang.

Corresponding author

Correspondence to Hongli Jing.

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Li, C., Wang, L., Wu, N. et al. A retrospective study of bone scintigraphy in the follow-up of patients with synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome: is it useful to repeat bone scintigraphy for disease assessment?. Clin Rheumatol 39, 1305–1314 (2020). https://doi.org/10.1007/s10067-019-04864-z

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  • DOI: https://doi.org/10.1007/s10067-019-04864-z

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