Skip to main content

Advertisement

SpringerLink
Log in

SAPHO syndrome associated spondylitis

  • Original Article
  • Published:
European Spine Journal Aims and scope Submit manuscript

Abstract

The concept of synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome has been well clarified, after Chamot et al. suggested this peculiar disorder in 1987. The most commonly affected site in SAPHO syndrome is the anterior chest, followed by the spine. However, the clinical course and taxonomic concept of SAPHO spinal lesions are poorly understood. This study was performed to analyze: (1) the detailed clinical course of spinal lesions in SAPHO syndrome, and (2) the relationship between SAPHO syndrome with spinal lesions and seronegative spondyloarthropathy. Thirteen patients with spondylitis in SAPHO syndrome were analyzed. The features of spinal lesions were a chronic onset with a slight inflammatory reaction, and slowly progressing non-marginal syndesmophytes at multi spinal levels, besides the coexistence of specific skin lesions. SAPHO syndrome, especially spinal lesions related to palmoplantar pustulosis, can be recognized as a subtype of seronegative spondyloarthropathy.

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

Access this article

Log in via an institution

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

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1
Fig. 2
Fig. 3
Fig. 4

Similar content being viewed by others

References

  1. Amital H, Applbaum YH, Aamar S, Daniel N, Rubinow A (2004) SAPHO syndrome treated with pamidronate: an open-label study of 10 patients. Rheumatology (Oxford) 43:658–661. doi:10.1093/rheumatology/keh149

    Article  CAS  Google Scholar 

  2. Amor B, Dougados M, Mijiyawa M (1990) Criteria of the classification of spondylarthropathies. Rev Rhum Mal Osteoartic 57:85–89

    PubMed  CAS  Google Scholar 

  3. Ashurst PJ (1964) Relapsing pustular eruptions of the hands and feet. Br J Dermatol 76:169–180. doi:10.1111/j.1365-2133.1964.tbl4502.x

    Article  PubMed  CAS  Google Scholar 

  4. Chamot AM, Benhamou CL, Kahn MF, Beraneck L, Kaplan G, Prost A (1987) Acne-pustulosis-hyperostosis-osteitis syndrome. Results of a national survey. 85 cases. Rev Rhum Mal Osteoartic 54:187–196

    PubMed  CAS  Google Scholar 

  5. Court C, Charlez C, Molina V, Clerc D, Miquel A, Nordin JY (2005) Isolated thoracic spine lesion: is this the presentation of a SAPHO syndrome? A case report. Eur Spine J 14:711–715. doi:10.1007/s00586-004-0791-4

    Article  PubMed  Google Scholar 

  6. Dougados M, van der Linden S, Juhlin R, Huitfeldt B, Amor B, Calin A, Cats A, Dijkmans B, Olivieri I, Pasero G et al (1991) The European spondylarthropathy study group preliminary criteria for the classification of spondylarthropathy. Arthritis Rheum 34:1218–1227. doi:10.1002/art 1780341003

    Article  PubMed  CAS  Google Scholar 

  7. Earwaker JW, Cotten A (2003) SAPHO: syndrome or concept? Imaging findings. Skeletal Radiol 32:311–327

    PubMed  CAS  Google Scholar 

  8. Edlund E, Johnsson U, Lidgren L, Pettersson H, Sturfelt G, Svensson B, Theander J, Willen H (1998) Palmoplantar pustulosis and sternocostoclavicular arthro-osteitis. Ann Rheum Dis 47:809–815

    Article  Google Scholar 

  9. Gerster JC, Lagier R, Livio JJ (1990) Propionibacterium acnes in a spondylitis with palmoplantar pustulosis. Ann Rheum Dis 49:337–338

    Article  PubMed  CAS  Google Scholar 

  10. Guri J (1946) Pyogenic osteomyelitis of spine. J Bone Joint Surg 28:29–39

    Google Scholar 

  11. Hayem G, Bouchaud-Chabot A, Benali K, Roux S, Palazzo E, Silbermann-Hoffman O, Kahn MF, Meyer O (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

    Article  PubMed  CAS  Google Scholar 

  12. Higashi S, Nakamura T, Tomoda K, Tsukano M, Sakaguchi M, Kuga F (2005) A study of ten Japanese patients with seronegative spondylarthropathy: a tentative proposal. Mod Rheumatol 15:329–335. doi:10.1007/s 10165-005-0414-x

    Article  PubMed  Google Scholar 

  13. Hukuda S, Minami M, Saito T, Mitsui H, Matsui N, Komatsubara Y, Makino H, Shibata T, Shingu M, Sakou T, Shichikawa K (2001) Spondyloarthropathies in Japan: nationwide questionnaire survey performed by the Japan Ankylosing Spondylitis Society. J Rheumatol 28:554–559

    PubMed  CAS  Google Scholar 

  14. Jurik AG, Graudal H, de Carvalho A (1985) Sclerotic changes of the manubrium sterni. Skeletal Radiol 13:195–201. doi:10.1007/BF00350573

    Article  PubMed  CAS  Google Scholar 

  15. Kahn MF, Bouvier M, Palazzo E, Tebib JG, Colson F (1991) Sternoclavicular pustulotic osteitis (SAPHO). 20-year interval between skin and bone lesions. J Rheumatol 18:1104–1108

    PubMed  CAS  Google Scholar 

  16. Khan MA (2003) Clinical features of ankylosing spondylitis. In: Hochberg MC, Silman AJ, Smolen JS, Weinblatt ME, Weisman MH (eds) Rheumatology. Mosby, St. Louis, pp 1161–1181

    Google Scholar 

  17. Kotilainen P, Merilahti-Palo R, Lehtonen OP, Manner I, Helander I, Möttönen T, Rintala E (1996) Propionibacterium acnes isolated from sternal osteitis in a patient with SAPHO syndrome. J Rheumatol 23:1302–1304

    PubMed  CAS  Google Scholar 

  18. Kotilainen P, Gullichsen RE, Saario R, Manner I, Kotilainen E (1997) Aseptic spondylitis as the initial manifestation of the SAPHO syndrome. Eur Spine J 6:327–329. doi:10.1007/BF01142679

    Article  PubMed  CAS  Google Scholar 

  19. Kulowski J (1936) Pyogenic osteomyelitis of the spine. An analysis and discussion of 102 cases. J Bone Joint Surg 18:343–364

    Google Scholar 

  20. Laredo JD, Vuillemin-Bodaghi V, Boutry N, Cotten A, Parlier-Cuau C (2007) SAPHO Syndrome: MR Appearance of Vertebral Involvement. Radiology 242:825–831. doi:10.1148/radiol 2423051222

    Article  PubMed  Google Scholar 

  21. Maugars Y, Berthelot JM, Ducloux JM, Prost A (1995) SAPHO syndrome: a followup study of 19 cases with special emphasis on enthesis involvement. J Rheumatol 22:2135–2141

    PubMed  CAS  Google Scholar 

  22. McEwen C, DiTata D, Lingg C, Porini A, Good A, Rankin T (1971) Ankylosing spondylitis and spondylitis accompanying ulcerative colitis, regional enteritis, psoriasis and Reiter’s disease. A comparative study. Arthritis Rheum 14:291–318. doi:10.1002/art.1780140302

    Article  PubMed  CAS  Google Scholar 

  23. Moll C, Hernández MV, Cañete JD, Gómez-Puerta JA, Soriano A, Collado A, Sanmartí R (2008) Ilium osteitis as the main manifestation of the SAPHO syndrome: response to infliximab therapy and review of the literature. Semin Arthritis Rheum 37:299–306. doi:10.1016/j.semarthrit.2007.08.004

    Article  PubMed  CAS  Google Scholar 

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

    Google Scholar 

  25. Sonozaki H, Kawashima M, Hongo O, Yaoita H, Ikeno M, Matsuura M, Okai K, Azuma A (1981) Incidence of arthro-osteitis in patients with pustulosis palmaris et plantaris. Ann Rheum Dis 40:554–557

    Article  PubMed  CAS  Google Scholar 

  26. Sonozaki H, Mitsui H, Miyanaga Y, Okitsu K, Igarashi M, Hayashi Y, Matsuura M, Azuma A, Okai K, Kawashima M (1981) Clinical features of 53 cases with pustulotic arthro-osteitis. Ann Rheum Dis 40:547–553

    Article  PubMed  CAS  Google Scholar 

  27. Sugimoto H, Tamura K, Fujii T (1998) The SAPHO syndrome: defining the radiologic spectrum of diseases comprising the syndrome. Eur Radiol 8:800–806. doi:10.1007/s003300050475

    Article  PubMed  CAS  Google Scholar 

  28. Takigawa T, Tanaka M, Nakahara S, Sugimoto Y, Ozaki T (2008), SAPHO syndrome with rapidly progressing destructive spondylitis: two cases treated surgically. Eur Spine J. doi:10.1007/s00586-008-0663-4

  29. Toussirot E, Dupond JL, Wendling D (1997) Spondylodiscitis in SAPHO syndrome. A series of eight cases. Ann Rheum Dis 56:52–58

    Article  PubMed  CAS  Google Scholar 

Download references

Acknowledgments

No funds were received in support of this work.

Author information

Authors and Affiliations

  1. Department of Orthopaedic Surgery, Okayama University Hospital, 2-5-1 Shikata-cho, Okayama City, Okayama, 700-8558, Japan

    Tomoyuki Takigawa, Masato Tanaka, Kazuo Nakanishi, Haruo Misawa, Yoshihisa Sugimoto, Tomohiro Takahata, Hiroyuki Nakahara & Toshifumi Ozaki

  2. Department of Orthopaedic Surgery, National Hospital Organization Okayama Medical Center, 1711-1 Tamasu, Okayama City, Okayama, 701-1192, Japan

    Shinnosuke Nakahara

Authors
  1. Tomoyuki Takigawa
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Masato Tanaka
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Kazuo Nakanishi
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Haruo Misawa
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Yoshihisa Sugimoto
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. Tomohiro Takahata
    View author publications

    You can also search for this author in PubMed Google Scholar

  7. Hiroyuki Nakahara
    View author publications

    You can also search for this author in PubMed Google Scholar

  8. Shinnosuke Nakahara
    View author publications

    You can also search for this author in PubMed Google Scholar

  9. Toshifumi Ozaki
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Tomoyuki Takigawa.

Rights and permissions

Reprints and permissions

About this article

Cite this article

Takigawa, T., Tanaka, M., Nakanishi, K. et al. SAPHO syndrome associated spondylitis. Eur Spine J 17, 1391–1397 (2008). https://doi.org/10.1007/s00586-008-0722-x

Download citation

  • Received:

  • Revised:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00586-008-0722-x

Keywords

Search

Navigation