Skip to main content

Advertisement

Log in

Mucosal lesions may be a minor complication of SAPHO syndrome: a study of 11 Japanese patients with SAPHO syndrome

  • Original Article
  • Published:
Rheumatology International Aims and scope Submit manuscript

Abstract

Since the term synovitis–acne–pustulosis–hyperostosis–osteitis (SAPHO) syndrome was proposed by Chamot et al. (Rev Rhum Mal Osteoartic 54:187–196, 1987), clinical reviews concerning this syndrome have been mainly reported from Europe. We carried out a retrospective analysis of 11 Japanese patients with SAPHO syndrome, and reviewed the clinical features of our series in comparison with those in a European large case study. In this study the major features of SAPHO syndrome were chronic osteitis of the anterior chest wall and pustulotic arthro-ostitis with middle age onset, and mucosal lesions seemed to be a minor complication of SAPHO syndrome. The non-erosive peripheral large joints arthritis and the particular HLA types (HLA-B51, B52, or A26), which had been reported to be increased in Behcet’s disease, were frequently seen in SAPHO syndrome with mucosal lesions. This study also suggests that SAPHO syndrome with mucosal lesions may be part of a broader disease spectrum including Behcet’s disease.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Fig. 1
Fig. 2

Similar content being viewed by others

References

  1. 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

    CAS  PubMed  Google Scholar 

  2. Chigira M, Shimizu T (1989) Computed tomographic appearances of sternocostoclavicular hyperostosis. Skeletal Radiol 18:347–352

    Article  CAS  PubMed  Google Scholar 

  3. 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

    Article  CAS  PubMed  Google Scholar 

  4. Sonozaki H, Mitsui H, Miyanaga Y et al (1981) Clinical features of 53 cases with pustulotic arthro-osteitis. Ann Rheum Dis 40:547–553

    Article  CAS  PubMed  Google Scholar 

  5. Knitzer RH, Needleman BW (1991) Musculoskeletal syndromes associated with acne. Semin Arthritis Rheum 20:247–255

    Article  CAS  PubMed  Google Scholar 

  6. Laasonen LS, Karvonen SL, Reunala TL (1994) Bone disease in adolescents with acne fulminans and severe cystic acne: radiologic and scintigraphic finding. AJR Am J Roentgenol 162:1161–1165

    CAS  PubMed  Google Scholar 

  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

    Article  CAS  PubMed  Google Scholar 

  8. 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

    CAS  PubMed  Google Scholar 

  9. 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

    Article  CAS  Google Scholar 

  10. Yamaguchi A, Tsuchiya N, Mitsui H (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

    Article  CAS  PubMed  Google Scholar 

  11. Arber N, Klein T, Meiner Z (1991) Close association of HLA-B51 and B52 in Israeli patients with Behcet’s syndrome. Ann Rheum Dis 50:351–353

    Article  CAS  PubMed  Google Scholar 

  12. Chung YM, Yeh TS, Sheu MM et al (1990) Behcet’s disease with ocular involvement in Taiwan: a joint survey of six major ophthalmological departments. J Formos Med Assoc 89:413–417

    CAS  PubMed  Google Scholar 

  13. Kamiishi T, Itoh Y, Meguro A et al (2008) Four-digit allele genotyping of HLA-A and HLA-B genes in Japanese patients with Behçet’s disease (BD) by a PCR-SSOP-luminex method and stratification analysis according to each major symptom of BD. Jpn J Ophthalmol 112:451–458

    CAS  Google Scholar 

  14. Mizuki N, Ohno S, Ando H (1997) A strong association between HLA-B*5101 and Behcet’s disease in Greek patients. Tissue Antigens 50:57–60

    Article  CAS  PubMed  Google Scholar 

  15. Sakane T, Takeno M, Suzuki N et al (1999) Behcet’s disease. N Engl J Med 341:1284–1291

    Article  CAS  PubMed  Google Scholar 

  16. Sasaki T (1967) A case with osteomyelitis of the bilateral clavicles association with pustulosis palmaris et plantaris. Rinsho Seikeigeka 2:333–337

    Google Scholar 

  17. Colina M, Govoni M, Orzincolo C et al (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  PubMed  Google Scholar 

  18. Mejjad O, Daragon A, Louvel JP et al (1996) Osteoarticular manifestations of pustulosis palmaris et plantaris and of psoriasis: two distinct entities. Ann Rheum Dis 55:177–180

    Article  CAS  PubMed  Google Scholar 

  19. Queiro R, Moreno P, Sarasqueta C et al (2008) Synovitis–acne–pustulosis–hyperostosis–osteitis syndrome and psoriatic arthritis exhibit a different immunogenetic profile. Clin Exp Rheumatol 26:125–128

    CAS  PubMed  Google Scholar 

  20. Shinohara Y (1996) Clinical and radiological study on sterno-cost-clavicular hyperostosis. J Med Soc Toho Univ 34:8–363

    Google Scholar 

  21. Wagner AD, Andresen J, Jendro MC et al (2002) Sustained response to tumor necrosis factor alpha-blocking agents in two patients with SAPHO syndrome. Arthritis Rheum 46:1965–1968

    Article  PubMed  Google Scholar 

  22. Massara A, Cavazzini PL, Trotta F (2006) In SAPHO syndrome anti-TNF-alpha therapy may induce persistent amelioration of osteoarticular complaints, but may exacerbate cutaneous manifestations. Rheumatology (Oxford) 45:730–733

    Article  CAS  Google Scholar 

  23. Caravatti M, Wiesli P, Uebelhart D et al (2002) Coincidence of Behcet’s disease and SAPHO syndrome. Clin Rheumatol 21:324–327

    Article  CAS  PubMed  Google Scholar 

  24. 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

    Article  CAS  Google Scholar 

  25. Yabe H, Takano Y, Nomura E et al (2008) Two cases of SAPHO syndrome accompanied by classic features of Behcet’s disease and review of the literature. Clin Rheumatol 27:133–135

    Article  PubMed  Google Scholar 

  26. James DG (1986) Silk route disease. Postgrad Med J 62:151–153

    Article  CAS  PubMed  Google Scholar 

  27. Mason RM, Barnes CG (1969) Behcet’s syndrome with arthritis. Ann Rheum Dis 28:95–103

    Article  CAS  PubMed  Google Scholar 

  28. Nanke Y, Kotake S, Momohara S et al (2002) Synovial histology in three Behcet’s disease patients with orthopedic surgery. Clin Exp Rheumatol 20:S35–S39

    CAS  PubMed  Google Scholar 

  29. Diri E, Mat C, Hamuryudan V et al (2001) Papulopustular skin lesions are seen more frequently in patients with Behcet’s syndrome who have arthritis: a controlled and masked study. Ann Rheum Dis 60:1074–1076

    Article  CAS  PubMed  Google Scholar 

  30. Orui H, Takahara M, Ishikawa A et al (2002) Radiological features of long bones in synovitis, acne, pustulosis, hyperostosis, osteitis syndrome and their correlation with pathological findings. Mod Rheumatol 12:56–63

    Google Scholar 

  31. Kitano M, Hasegawa M, Kinoshita Y et al (2006) Gonarthrosis with pustulosis palmaris et plantaris: a case report. Kanto J Orthop Traumatol 37:8–12

    Google Scholar 

Download references

Acknowledgments

The authors thank Dr. Kenichiro Matsuzaki, Dr. Masaaki Takahashi, Dr. Akio Yokoi (Department of Orthopaedic Surgery, National Hospital Organization Tokyo Medical Center), and Dr. Eiki Nomura (Department of Orthopaedic Surgery, Saitama Municipal Hospital) for their helpful discussion.

Conflict of interest statement

All authors have no conflict of interest to declare.

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Hiroki Yabe.

Rights and permissions

Reprints and permissions

About this article

Cite this article

Yabe, H., Ohshima, H., Takano, Y. et al. Mucosal lesions may be a minor complication of SAPHO syndrome: a study of 11 Japanese patients with SAPHO syndrome. Rheumatol Int 30, 1277–1283 (2010). https://doi.org/10.1007/s00296-009-1138-6

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00296-009-1138-6

Keywords

Navigation