Abstract
We describe two patients with SAPHO (synovitis–acne–pustulosis–hyperostosis–ostitis) syndrome who presented some of the classic features of Behcet’s disease. The first case is a man diagnosed as SAPHO at 74 years old. His major complaint is pain and swelling of the bilateral sterno-clavicular region for more than 14 years. Another conspicuous complication was bilateral glaucoma and episodes of iritis were recognized during the follow-up period. The second case is a 65-year-old woman, who first consulted us with right knee pain. As she had a past history of palmoplantar pustulosis and anterior chest pain, her sterile knee arthritis was diagnosed as SAPHO. She also had been suffering from recurrent oral aphthous ulceration since 6 months before visiting our hospital. Considering the clinical courses of our two cases and a review of five previously reported cases, these conditions may imply that classic features of Behcet’s disease are minor complications of SAPHO syndrome. Human leukocyte antigen typing and frequent association of sacroiliitis in our cases and in the review of the literature for SAPHO syndrome with some of the classic features of Behcet’s disease may indicate this condition to be a closely related disease with seronegative spondylo-arthritis.
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References
Chamot AM, Benhamou CL, Kahn MF et al (1987) Acne-pustulosis-hyperostosis-osteitis syndrome. Results of a national survey. 85 cases. Rev Rhum Mal Osteoartic 54:187–196
Kahn MF (1993) Psoriatic arthritis and synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome. Curr Opin Rheumatol 5:428–435
Sonozaki H, Azuma A, Okai K et al (1979) Clinical features of 22 cases with “inter-sterno-costo-clavicular ossification”. A new rheumatic syndrome. Arch Orthop Trauma Surg 95:13–22
Chigira M, Shimizu T (1989) Computed tomographic appearances of sternocostoclavicular hyperostosis. Skeletal Radiol 18:347–352
Behcet HH (1937) Uber rezidivierende aphtose, dorch ein virus verursachte Geschwuren am Mund, am Auge und an den Genitalen. Dermatol Wochenschr 105:1152–1157
Jorizzo JL (1987) Behcet’s disease. Neurol Clin 5:427–40
Terashima K, Shirato M, Nishimaki T et al (1988) A case report of Behcet disease of incomplete type accompanied by pustulotic arthro-osteitis. Ryumachi 28:34–40
Villaverde V, Munnoz-Fernandez S, Hidalgo V et al (1999) Acute anterior uveitis in a patient with sacroiliitis and acne conglobata. Rheumatology (Oxford) 38:797–798
Caravatti M, Wiesli P, Uebelhart D et al (2002) Coincidence of Behcet’s disease and SAPHO syndrome. Clin Rheumatol 21:324–327
Yamasaki O, Iwatsuki K, Kaneko F (2003) A case of SAPHO syndrome with pyoderma gangrenosum and inflammatory bowel disease masquerading as Behcet’s disease. Adv Exp Med Biol 528:339–341
Yamada R, Komatsu A, Sujino T et al (1994) A case of sternocostclavicular hyperostosis with uveitis and secondary glaucoma. Jpn J Clin Ophthalmol 48:309–312
Yamaguchi A, Tsuchiya N, Mitsui H et al (1995) Association of HLA-B39 with HLA-B27-negative ankylosing spondylitis and pauciarticular juvenile rheumatoid arthritis in Japanese patients. Evidence for a role of the peptide-anchoring B pocket. Arthritis Rheum 38:1672–1677
Kinouchi Y, Matsumoto K, Negoro K et al (2003) Hla-B genotype in Japanese patients with Crohn’s disease. Dis Colon Rectum 46:S10–S14
Wei JC, Tsai WC, Lin HS et al (2004) HLA-B60 and B61 are strongly associated with ankylosing spondylitis in HLA-B27-negative Taiwan Chinese patients. Rheumatology (Oxford) 43:839–842
Higashi S, Nakamura T, Tomoda K et al (2005) A study of ten Japanese patients with seronegative spondylarthropathy: a tentative proposal. Mod Rheumatol 15:329–335
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Yabe, H., Takano, Y., Nomura, E. et al. Two cases of SAPHO syndrome accompanied by classic features of Behcet’s disease and review of the literature. Clin Rheumatol 27, 133–135 (2008). https://doi.org/10.1007/s10067-007-0697-8
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DOI: https://doi.org/10.1007/s10067-007-0697-8