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Published in: Journal of Orthopaedic Surgery and Research 1/2018

Open Access 01-12-2018 | Research article

F-18 FDG PET/CT in 26 patients with SAPHO syndrome: a new vision of clinical and bone scintigraphy correlation

Authors: Xiaochuan Sun, Chen Li, Yihan Cao, Ximin Shi, Li Li, Weihong Zhang, Xia Wu, Nan Wu, Hongli Jing, Wen Zhang

Published in: Journal of Orthopaedic Surgery and Research | Issue 1/2018

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Abstract

Backgrounds

Whole-body bone scintigraphy (WBBS) and MRI are widely used in assessment of patients with synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome. However, the value of F-18 fluorodeoxyglucose-positron emission tomography/computed tomography (18F-FDG PET/CT) in SAPHO syndrome was unclear. The aim of this study was to characterize the manifestation of SAPHO syndrome on 18F-FDG PET/CT and explore its relationship with clinical symptoms and WBBS.

Methods

Twenty-six patients who suffered from SAPHO syndrome and had undergone whole-body 18F-FDG PET/CT were recruited in Peking Union Medical College Hospital from 2004 to 2016. Clinical manifestations and laboratory findings were recorded for all patients. Imaging data on 18F-FDG PET/CT and WBBS were collected and analyzed retrospectively.

Results

All the 26 patients (20 females and 6 males) exhibited skeletal abnormalities on 18F-FDG PET/CT. Multiple skeletal lesions affecting the anterior chest wall or spine with low to moderate 18F-FDG uptake and coexistence of osteolysis and osteosclerosis presented as the typical features of SAPHO syndrome. Sixteen (61.5%) patients had abnormal 18F-FDG uptake outside the osteoarticular system. PET scan had moderate to substantial agreement with CT and WBBS in revealing lesions in the anterior chest wall and axial skeleton. Nonetheless, the correlation between increased 18F-FDG uptake and clinical symptoms was weak.

Conclusions

SAPHO syndrome exhibits characteristic features on 18F-FDG PET/CT. It showed comparable capacity in revealing skeletal lesions with bone scintigraphy.
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Literature
1.
go back to reference Chamot A, Benhamou C, Kahn M, Beraneck L, Kaplan G, Prost A. Acne-pustulosis-hyperostosis-osteitis syndrome. Results of a national survey. 85 cases. Revue du rhumatisme et des maladies ostéo-articulaires. 1987;54(3):187–96.PubMed Chamot A, Benhamou C, Kahn M, Beraneck L, Kaplan G, Prost A. Acne-pustulosis-hyperostosis-osteitis syndrome. Results of a national survey. 85 cases. Revue du rhumatisme et des maladies ostéo-articulaires. 1987;54(3):187–96.PubMed
3.
go back to reference Schaub S, Sirkis HM, Kay J. Imaging for synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome. Rheum Dis Clin N Am. 2016;42(4):695–710.CrossRef Schaub S, Sirkis HM, Kay J. Imaging for synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome. Rheum Dis Clin N Am. 2016;42(4):695–710.CrossRef
5.
go back to reference Magrey M, Khan MA. New insights into synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome. Curr Rheumatol Rep. 2009;11(5):329–33.CrossRefPubMed Magrey M, Khan MA. New insights into synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome. Curr Rheumatol Rep. 2009;11(5):329–33.CrossRefPubMed
6.
go back to reference Van Doornum S, Barraclough D, McColl G, Wicks I. SAPHO: rare or just not recognized? Semin Arthritis Rheum. 2000;30(1):70–7.CrossRefPubMed Van Doornum S, Barraclough D, McColl G, Wicks I. SAPHO: rare or just not recognized? Semin Arthritis Rheum. 2000;30(1):70–7.CrossRefPubMed
7.
go back to reference Witt M, Meier J, Hammitzsch A, Proft F, Schulze-Koops H, Grunke M. Disease burden, disease manifestations and current treatment regimen of the SAPHO syndrome in Germany: results from a nationwide patient survey. Semin Arthritis Rheum. 2014;43(6):745–50.CrossRefPubMed Witt M, Meier J, Hammitzsch A, Proft F, Schulze-Koops H, Grunke M. Disease burden, disease manifestations and current treatment regimen of the SAPHO syndrome in Germany: results from a nationwide patient survey. Semin Arthritis Rheum. 2014;43(6):745–50.CrossRefPubMed
9.
go back to reference Xu W, Li C, Zhao X, et al. Whole-spine computed tomography findings in SAPHO syndrome. J Rheumatol. 2017;44(5):648–54.CrossRefPubMed Xu W, Li C, Zhao X, et al. Whole-spine computed tomography findings in SAPHO syndrome. J Rheumatol. 2017;44(5):648–54.CrossRefPubMed
10.
go back to reference Kohlfuerst S, Igerc I, Lind P. FDG PET helpful for diagnosing SAPHO syndrome. Clin Nucl Med. 2003;28(10):838–9.CrossRefPubMed Kohlfuerst S, Igerc I, Lind P. FDG PET helpful for diagnosing SAPHO syndrome. Clin Nucl Med. 2003;28(10):838–9.CrossRefPubMed
11.
go back to reference Pichler R, Weiglein K, Schmekal B, Sfetsos K, Maschek W. Bone scintigraphy using Tc-99m DPD and F18-FDG in a patient with SAPHO syndrome. Scand J Rheumatol. 2003;32(1):58–60.CrossRefPubMed Pichler R, Weiglein K, Schmekal B, Sfetsos K, Maschek W. Bone scintigraphy using Tc-99m DPD and F18-FDG in a patient with SAPHO syndrome. Scand J Rheumatol. 2003;32(1):58–60.CrossRefPubMed
12.
go back to reference Shibakuki R, Seto T, Uematsu K, et al. Pulmonary adenocarcinoma associated with SAPHO syndrome difficult to differentiate from multiple bone metastasis. Intern Med. 2006;45(8):543–6.CrossRefPubMed Shibakuki R, Seto T, Uematsu K, et al. Pulmonary adenocarcinoma associated with SAPHO syndrome difficult to differentiate from multiple bone metastasis. Intern Med. 2006;45(8):543–6.CrossRefPubMed
13.
go back to reference Inoue K, Yamaguchi T, Ozawa H, et al. Diagnosing active inflammation in the SAPHO syndrome using 18FDG-PET/CT in suspected metastatic vertebral bone tumors. Ann Nucl Med. 2007;21(8):477–80.CrossRefPubMed Inoue K, Yamaguchi T, Ozawa H, et al. Diagnosing active inflammation in the SAPHO syndrome using 18FDG-PET/CT in suspected metastatic vertebral bone tumors. Ann Nucl Med. 2007;21(8):477–80.CrossRefPubMed
14.
go back to reference Takeuchi K, Matsusita M, Takagishi K. A case of SAPHO (synovitis-acne-pustulosis-hyperostosis-osteomyelitis) syndrome in which [18F]fluorodeoxyglucose positron emission tomography was useful for differentiating from multiple metastatic bone tumors. Mod Rheumatol. 2007;17(1):67–71.CrossRefPubMed Takeuchi K, Matsusita M, Takagishi K. A case of SAPHO (synovitis-acne-pustulosis-hyperostosis-osteomyelitis) syndrome in which [18F]fluorodeoxyglucose positron emission tomography was useful for differentiating from multiple metastatic bone tumors. Mod Rheumatol. 2007;17(1):67–71.CrossRefPubMed
15.
go back to reference Patel CN, Smith JT, Rankine JJ, Scarsbrook AF. F-18 FDG PET/CT can help differentiate SAPHO syndrome from suspected metastatic bone disease. Clin Nucl Med. 2009;34(4):254–7.CrossRefPubMed Patel CN, Smith JT, Rankine JJ, Scarsbrook AF. F-18 FDG PET/CT can help differentiate SAPHO syndrome from suspected metastatic bone disease. Clin Nucl Med. 2009;34(4):254–7.CrossRefPubMed
16.
go back to reference Abuhid IM, Silva LC, Martins GP, de Rezende NA. Diagnosing SAPHO syndrome in suspected metastatic bone tumors. Clin Nucl Med. 2010;35(3):172–4.CrossRefPubMed Abuhid IM, Silva LC, Martins GP, de Rezende NA. Diagnosing SAPHO syndrome in suspected metastatic bone tumors. Clin Nucl Med. 2010;35(3):172–4.CrossRefPubMed
17.
go back to reference Canbaz F, Gonullu G, Baris S, Selcuk MB, Bicakci N. SAPHO syndrome without dermatologic manifestations: multifocal uptake mismatch on 99mTc-MDP and 18FDG-FDG-PET/CT imaging. Hell J Nucl Med. 2010;13(1):73.PubMed Canbaz F, Gonullu G, Baris S, Selcuk MB, Bicakci N. SAPHO syndrome without dermatologic manifestations: multifocal uptake mismatch on 99mTc-MDP and 18FDG-FDG-PET/CT imaging. Hell J Nucl Med. 2010;13(1):73.PubMed
18.
go back to reference J-i N, Yamada K, Mitsugi N, Saito T. A case of SAPHO syndrome with destructive spondylodiscitis suspicious of tuberculous spondylitis. Mod Rheumatol. 2010;20(1):93–7.CrossRef J-i N, Yamada K, Mitsugi N, Saito T. A case of SAPHO syndrome with destructive spondylodiscitis suspicious of tuberculous spondylitis. Mod Rheumatol. 2010;20(1):93–7.CrossRef
20.
go back to reference Ikeda K, Yamagata M, Tanaka S, Yokota M, Furuta S, Nakajima H. Synovitis and osteitis in the left sternoclavicular joint in a 60-year-old woman. J Med Ultrason. 2015;42(1):133–4.CrossRef Ikeda K, Yamagata M, Tanaka S, Yokota M, Furuta S, Nakajima H. Synovitis and osteitis in the left sternoclavicular joint in a 60-year-old woman. J Med Ultrason. 2015;42(1):133–4.CrossRef
21.
go back to reference Dong A, Bai Y, Cui Y, Zhang J, Zuo C. FDG PET/CT in early and late stages of SAPHO syndrome: two case reports with MRI and bone scintigraphy correlation. Clin Nucl Med. 2016;41(4):e211–e5.CrossRefPubMed Dong A, Bai Y, Cui Y, Zhang J, Zuo C. FDG PET/CT in early and late stages of SAPHO syndrome: two case reports with MRI and bone scintigraphy correlation. Clin Nucl Med. 2016;41(4):e211–e5.CrossRefPubMed
22.
go back to reference Depasquale R, Kumar N, Lalam RK, et al. SAPHO: what radiologists should know. Clin Radiol. 2012;67(3):195–206.CrossRefPubMed Depasquale R, Kumar N, Lalam RK, et al. SAPHO: what radiologists should know. Clin Radiol. 2012;67(3):195–206.CrossRefPubMed
23.
go back to reference Cicchetti DV, Feinstein AR. High agreement but low kappa: II. Resolving the paradoxes. J Clin Epidemiol. 1990;43(6):551–8.CrossRefPubMed Cicchetti DV, Feinstein AR. High agreement but low kappa: II. Resolving the paradoxes. J Clin Epidemiol. 1990;43(6):551–8.CrossRefPubMed
24.
go back to reference Feinstein AR, Cicchetti DV. High agreement but low kappa: I. The problems of two paradoxes. J Clin Epidemiol. 1990;43(6):543–9.CrossRefPubMed Feinstein AR, Cicchetti DV. High agreement but low kappa: I. The problems of two paradoxes. J Clin Epidemiol. 1990;43(6):543–9.CrossRefPubMed
25.
go back to reference Colina M, Govoni M, Orzincolo C, Trotta F. Clinical and radiologic evolution of synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome: a single center study of a cohort of 71 subjects. Arthritis Care Res. 2009;61(6):813–21.CrossRef Colina M, Govoni M, Orzincolo C, Trotta F. Clinical and radiologic evolution of synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome: a single center study of a cohort of 71 subjects. Arthritis Care Res. 2009;61(6):813–21.CrossRef
26.
go back to reference Hayem G, Bouchaud-Chabot A, Benali K, et al. SAPHO syndrome: a long-term follow-up study of 120 cases. Semin Arthritis Rheum. 1999;29(3):159–71.CrossRefPubMed Hayem G, Bouchaud-Chabot A, Benali K, et al. SAPHO syndrome: a long-term follow-up study of 120 cases. Semin Arthritis Rheum. 1999;29(3):159–71.CrossRefPubMed
28.
go back to reference Li C, Zuo Y, Wu N, et al. Synovitis, acne, pustulosis, hyperostosis and osteitis syndrome: a single centre study of a cohort of 164 patients. Rheumatology. 2016;55(6):1023–30.CrossRefPubMed Li C, Zuo Y, Wu N, et al. Synovitis, acne, pustulosis, hyperostosis and osteitis syndrome: a single centre study of a cohort of 164 patients. Rheumatology. 2016;55(6):1023–30.CrossRefPubMed
29.
go back to reference McGauvran A, Kotsenas A, Diehn F, Wald J, Carr C, Morris J. SAPHO syndrome: imaging findings of vertebral involvement. Am J Neuroradiol. 2016;37(8):1567–72.CrossRefPubMed McGauvran A, Kotsenas A, Diehn F, Wald J, Carr C, Morris J. SAPHO syndrome: imaging findings of vertebral involvement. Am J Neuroradiol. 2016;37(8):1567–72.CrossRefPubMed
30.
go back to reference Anić B, Padjen I, Mayer M, Bosnić D, Cerovec M. Clinical features of the SAPHO syndrome and their role in choosing the therapeutic approach: report of four patients and review of the literature. Acta Dermatovenerol Croat. 2014;22(3):180–8.PubMed Anić B, Padjen I, Mayer M, Bosnić D, Cerovec M. Clinical features of the SAPHO syndrome and their role in choosing the therapeutic approach: report of four patients and review of the literature. Acta Dermatovenerol Croat. 2014;22(3):180–8.PubMed
31.
go back to reference Nguyen MT, Borchers A, Selmi C, Naguwa SM, Cheema G, Gershwin ME. The SAPHO syndrome. Semin Arthritis Rheum. 2012;42(3):254–65.CrossRefPubMed Nguyen MT, Borchers A, Selmi C, Naguwa SM, Cheema G, Gershwin ME. The SAPHO syndrome. Semin Arthritis Rheum. 2012;42(3):254–65.CrossRefPubMed
32.
go back to reference Fujii T, Matsudaira K, Oda H, Seichi A, Nakamura K. A case of SAPHO syndrome with paraplegia due to a thoracic kyphosis. Ryumachi. 2002;42(4):687–93.PubMed Fujii T, Matsudaira K, Oda H, Seichi A, Nakamura K. A case of SAPHO syndrome with paraplegia due to a thoracic kyphosis. Ryumachi. 2002;42(4):687–93.PubMed
33.
go back to reference Takigawa T, Tanaka M, Nakahara S, Sugimoto Y, Ozaki T. SAPHO syndrome with rapidly progressing destructive spondylitis: two cases treated surgically. Eur Spine J. 2008;17(2):331–7.CrossRefPubMedCentral Takigawa T, Tanaka M, Nakahara S, Sugimoto Y, Ozaki T. SAPHO syndrome with rapidly progressing destructive spondylitis: two cases treated surgically. Eur Spine J. 2008;17(2):331–7.CrossRefPubMedCentral
34.
go back to reference Yamamoto T. Pustulotic arthro-osteitis associated with palmoplantar pustulosis. J Dermatol. 2013;40(11):857–63.CrossRefPubMed Yamamoto T. Pustulotic arthro-osteitis associated with palmoplantar pustulosis. J Dermatol. 2013;40(11):857–63.CrossRefPubMed
35.
go back to reference Shimizu S, Yukawa K, Kawaguchi S, Okubo Y, Suzuki M. Fluctuating mixed-type hearing loss associated with synovitis–acne–pustulosis–hyperostosis–osteomyelitis (SAPHO) syndrome. Auris Nasus Larynx. 2010;37(4):508–10.CrossRefPubMed Shimizu S, Yukawa K, Kawaguchi S, Okubo Y, Suzuki M. Fluctuating mixed-type hearing loss associated with synovitis–acne–pustulosis–hyperostosis–osteomyelitis (SAPHO) syndrome. Auris Nasus Larynx. 2010;37(4):508–10.CrossRefPubMed
36.
go back to reference Shiraishi W, Hayashi S, Iwanaga Y, Murai H, Yamamoto A, Kira J. A case of synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome presenting with hypertrophic pachymeningitis. J Neurol Sci. 2015;349(1-2):229–31.CrossRefPubMed Shiraishi W, Hayashi S, Iwanaga Y, Murai H, Yamamoto A, Kira J. A case of synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome presenting with hypertrophic pachymeningitis. J Neurol Sci. 2015;349(1-2):229–31.CrossRefPubMed
37.
go back to reference Hurtado-Nedelec M, Chollet-Martin S, Nicaise-Roland P, et al. Characterization of the immune response in the synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome. Rheumatology. 2008;47(8):1160–7.CrossRefPubMed Hurtado-Nedelec M, Chollet-Martin S, Nicaise-Roland P, et al. Characterization of the immune response in the synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome. Rheumatology. 2008;47(8):1160–7.CrossRefPubMed
38.
go back to reference Grosjean C, Hurtado-Nedelec M, Nicaise-Roland P, et al. Prevalence of autoantibodies in SAPHO syndrome: a single-center study of 90 patients. J Rheumatol. 2010;37(3):639–43.CrossRefPubMed Grosjean C, Hurtado-Nedelec M, Nicaise-Roland P, et al. Prevalence of autoantibodies in SAPHO syndrome: a single-center study of 90 patients. J Rheumatol. 2010;37(3):639–43.CrossRefPubMed
Metadata
Title
F-18 FDG PET/CT in 26 patients with SAPHO syndrome: a new vision of clinical and bone scintigraphy correlation
Authors
Xiaochuan Sun
Chen Li
Yihan Cao
Ximin Shi
Li Li
Weihong Zhang
Xia Wu
Nan Wu
Hongli Jing
Wen Zhang
Publication date
01-12-2018
Publisher
BioMed Central
Published in
Journal of Orthopaedic Surgery and Research / Issue 1/2018
Electronic ISSN: 1749-799X
DOI
https://doi.org/10.1186/s13018-018-0795-0

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