Abstract
Synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome is a rare group of sterile, inflammatory osteoarticular disorders classically associated with skin lesions. It is occasionally associated with enteropathic disease such as ulcerative colitis. We present a 39-year-old patient with chronic ulcerative colitis who developed enteropathic SAPHO and responded well to pamidronate. We discuss the clinicopathological features with particular attention to bone pathology, and perform a literature review of this fascinating syndrome.
This is a preview of subscription content, log in via an institution to check access.
References
Chamot AM et al (1987) Le syndrome acnè pustulose hyperostose ostèite (SAPHO)—Resultats d’une enquète nationale. Rev Rheumat 54:187–196
Kahn MF, Khan MA (1994) The SAPHO-syndrome. Baillieres Clin Rheumatol 8(2):333–362. doi:10.1016/S0950-3579(94)80022-7
Hayem G, Bouchaud-Chabot A, Benali K et al (1999) SAPHO syndrome: a long-term follow-up study of 120 cases. Semin Arthritis Rheum 29:159–171. doi:10.1016/S0049-0172(99)80027-4
Kotilainen P, Merilahti-Palo R, Lehtonen OP et al (1996) Propionibacterium acnes isolated from sternal osteitis in a patient with SAPHO syndrome. J Rheumatol 23(7):1302–1304
Steinoff JP, Cilursu A, Falasa G et al (2002) A study of musculoskeletal manifestations in 12 patients with SAPHO syndrome. J Clin Rheumatol 8:13–22. doi:10.1097/00124743-200202000-00005
Benhamou CL, Chamot AM, Kahn MF (1988) Synovitis-acne-pustulosis-hyperostosis-osteomyelitis syndrome (SAPHO): a new syndrome among the spondyloarthropathies? Clin Exp Rheumatol 6:109–112
Kahn MF, Bouchon JP, Chamot AM, Palazzo E (1992) Enterocolopathies chroniques et syndrome SAPHO. Rev Rheum Mal Osteoartic 59:91–94
Schilling F, Marker-Hermann E (2003) Chronic recurrent multifocal osteomyelitis in association with chronic inflammatory bowel disease: enteropathic CRMO. Z Rheumatol 62:527–538. doi:10.1007/s00393-003-0526-7
Bousvaros A, Marcon M, Treem W et al (1999) Chronic recurrent multifocal osteomyelitis associated with chronic inflammatory bowel disease in children. Dig Dis Sci 44:2500–2507. doi:10.1023/A:1026695224019
Schilling F (2004) SAPHO syndrome. Orphanet Encyclopedia. http://www.orpha.net/data/patho/GB/uk-sapho.pdf. Accessed 17 Feb 2009
Schilling F, Kessler S (2000) The SAPHO syndrome—clinical and radiological differentiation and classification on the basis of 86 cases. Rheumatology 59:1–28. doi:10.1007/s003930050001
Reith JD, Bauer TW, Schils JP (1996) Osseous manifestations of SAPHO (synovitis, acne, pustulosis, hyperostosis, osteitis) syndrome. Am J Surg Pathol 20:1368–1377. doi:10.1097/00000478-199611000-00008
Kahn MF, Chamot AM (1992) SAPHO syndrome. Rheum Dis Clin North Am 18:225–246
Schilling F, Wagner AD (2000) Azithromycin: an anti-inflammatory effect in chronic recurrent multifocal osteomyelitis? A preliminary report. Z Rheumatol 59(5):352–353. doi:10.1007/s003930070059
Amital H, Applbaum YH, Aamar S et al (2004) SAPHO syndrome treated with pamidronate: an open-label study of 10 patients. Rheumatology 43(5):658–661. doi:10.1093/rheumatology/keh149
Author information
Authors and Affiliations
Corresponding author
Rights and permissions
About this article
Cite this article
Siau, K., Laversuch, C.J. SAPHO syndrome in an adult with ulcerative colitis responsive to intravenous pamidronate: a case report and review of the literature. Rheumatol Int 30, 1085–1088 (2010). https://doi.org/10.1007/s00296-009-1021-5
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00296-009-1021-5