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Kim, Bae, Choi, Lee, Ji, and Song: A Case of Refractory SAPHO Syndrome Treated with Etanercept
Case Report
Journal of Rheumatic Diseases

Journal of Rheumatic Diseases 2012; 19(1): 51-54.

Published online: 28 February 2012

DOI: https://doi.org/10.4078/jrd.2012.19.1.51

A Case of Refractory SAPHO Syndrome Treated with Etanercept

Ye Ji Kim, Song I Bae, Sung Jae Choi, Young Ho Lee, Jong Dae Ji, Gwan Gyu Song

Division of Rheumatology, Department of Internal Medicine, College of Medicine, Korea University, Seoul, Korea.

Corresponding author (csjmd@hotmail.com)

Received 5 August 2011       Revised 17 August 2011       Accepted 17 August 2011

Copyright © 2012 Korean College of Rheumatology

Abstract

SAPHO syndrome, which has different skin changes and osteoarticular inflammation, is an acronym that stands for synovitis, acne, pustulosis, hyperostosis, and osteitis. Treatment of SAPHO syndrome includes non-steroidal anti-inflammatory drugs (NSAIDs), anti-rheumatic drugs, such as colchicines, corticosteroids and bisphosphonates, and disease-modifying agents. However, the treatment of SAPHO syndrome is controversial because it is a new clinical entity with unclear etiopathogenesis and inadequate clinical studies. We report a case with refractory SAPHO syndrome, which was successfully treated with a tumor necrosis factor (TNF)-α blocker.

Keywords: SAPHO syndrome, Treatment, TNF-α blocker

Figures and Tables

Figure 1
Acne in different size and shape were seen in his face.
jrd-19-51-g001
Figure 2
Coronal FSE fat saturated T2 weighted image showed irregularity of joint space and subchondral bone marrow edema and enhancement at the sacroiliac joint (arrow). These findings suggest sacroilitis.
jrd-19-51-g002
Figure 3
Technetium-99m bone scan shows increased uptake in the sternocleidoclavicular regions. This is known as "bull's head" or "bull-horn" sign and is characteristic for SAPHO syndrome.
jrd-19-51-g003

References

1. Olivieri I, Padula A, Ciancio G, Salvarani C, Niccoli L, Cantini F. Successful treatment of SAPHO syndrome with infliximab: report of two cases. Ann Rheum Dis. 2002. 61:375–376.
2. Matzaroglou Ch, Velissaris D, Karageorgos A, Marangos M, Panagiotopoulos E, Karanikolas M. SAPHO Syndrome Diagnosis and Treatment: Report of Five Cases and Review of the Literature. Open Orthop J. 2009. 3:100–106.
3. Hayem G, Bouchaud-Chabot A, Benali K, Roux S, Palazzo E, Silbermann-Hoffman O, et al. SAPHO syndrome: a long-term follow-up study of 120 cases. Semin Arthritis Rheum. 1999. 29:159–171.
4. Cotten A, Flipo RM, Mentre A, Delaporte E, Duquesnoy B, Chastanet P. SAPHO syndrome. Radiographics. 1995. 15:1147–1154.
5. Eyigor S, Karapolat H, Adanur H, Kirazli Y. SAPHO syndrome with adrenal deficiency: a case report. Cases J. 2009. 2:6281.
6. Hayem G. Valuable lessons from SAPHO syndrome. Joint Bone Spine. 2007. 74:123–126.
7. Van Doornum S, Barraclough D, McColl G, Wicks I. SAPHO: rare or just not recognized? Semin Arthritis Rheum. 2000. 30:70–77.
8. Chamot AM, Benhamou CL, Kahn MF, Beraneck L, Kaplan G, Prost A. Acne-pustulosis-hyperostosis-osteitis syndrome. Results of a national survey. 85 cases. Rev Rhum Mal Osteoartic. 1987. 54:187–196.
9. Assmann G, Kueck O, Kirchhoff T, Rosenthal H, Voswinkel J, Pfreundschuh M, et al. Efficacy of antibiotic therapy for SAPHO syndrome is lost after its discontinuation: an interventional study. Arthritis Res Ther. 2009. 11:R140.
10. Schwartz BD. Infectious agents, immunity, and rheumatic diseases. Arthritis Rheum. 1990. 33:457–465.
11. Duncan RC, Hay EM, Saklatvala J, Croft PR. Prevalence of radiographic osteoarthritis--it all depends on your point of view. Rheumatology (Oxford). 2006. 45:757–760.
12. Ben Abdelghani K, Dran DG, Gottenberg JE, Morel J, Sibilia J, Combe B. Tumor necrosis factor-alpha blockers in SAPHO syndrome. J Rheumatol. 2010. 37:1699–1704.
13. Vilar-Alejo J, Dehesa L, de la Rosa-del Rey P, Novoa-Medina J, Valerón Almazán P, Santana Medina N, et al. SAPHO syndrome with unusual cutaneous manifestations treated successfully with etanercept. Acta Derm Venereol. 2010. 90:531–532.
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