European Spine Journal

, Volume 6, Issue 5, pp 327–329 | Cite as

Aseptic spondylitis as the initial manifestation of the SAPHO syndrome

  • P. Kotilainen
  • R. E. Gullichsen
  • R. Saario
  • I. Manner
  • E. Kotilainen
Case Report


We describe the case of a 61-year-old female patient who presented with spondylitis of the lumbar spine. Although the microbiological cultures of the bone biopsy specimens obtained during laminotomy remained negative, the patient was treated with broad-spectrum antimicrobials for 2 months. Eight months later she started to suffer from pain and tenderness in her sternum and the medial portion of her left clavicle. The findings of computed tomography and gallium-labelled isotope scan were indicative of sternoclavicular arthritis. Again, all surgically obtained biopsy specimens yielded negative results in microbiological studies. The diagnosis of the SAPHO (synovitis, acne, pustulosis, hyperostosis, and osteomyelitis) syndrome was then made based on the clinical presentation with recurrent sterile osteitis in two characteristic locations, and the patient was started on immunosuppressive therapy. This case is a reminder that SAPHO may sometimes occur without any skin manifestations. Since this type of patient may be admitted to an orthopedic ward, it is important that orthopedic surgeons are familiar with the syndrome.

Key words

SAPHO Aseptic spondylitis 


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  1. 1.
    Benhamou CL, Enjolras O, Delrieu F, Prier A, Camus JP, Amor B (1984) Arthrites pseudo-septiques et bacterides d'Andrews. Rev Rhum Mal Osteoartic 51:229–231Google Scholar
  2. 2.
    Benhamou CL, Chamot AM, Kahn MF (1988) Synovitis-acne-pustulosis hyperostosis-osteomyelitis syndrome (Sapho). A new syndrome among the spondyloarthropathies? Clin Exp Rheumatol 6:109–112Google Scholar
  3. 3.
    Björksten B, Gustavson KH, Eriksson B, Lindholm Å, Nordström S (1978) Chronic recurrent multifocal osteomyelitis and pustulosis palmoplantaris. J Pediatr 93:227–231Google Scholar
  4. 4.
    Brooks GF, Pons VG (1994) Osteomyelitis. In: Hoeprich PD, Jordan MC, Ronald AR (eds) Infectious diseases. Lippincott, Philadelphia; pp 1390–1401Google Scholar
  5. 5.
    Ellis BI, Shier CK, Leisen JJ, Kastan DJ, McGoey JW (1987) Acne-associated spondylarthropathy: radiographic features. Radiology 162:541–545Google Scholar
  6. 6.
    Fallet GH, Arroyo J, Vischer TL (1983) Stemocostoclavicular hyperostosis: case report with a 31-year followup. Arthritis Rheum 26:784–790Google Scholar
  7. 7.
    Goossens M, Vanderstraeten C, Claessens H (1985) Sternocostoclavicular hyperostosis. A case report and review of the literature. Clin Orthop 194:164–168Google Scholar
  8. 8.
    Jurik AG, Helmig O, Graudal H (1988) Skeletal disease, arthro-osteitis, in adult patients with pustulosis palmoplantaris. Scand J Rheumatol [Suppl] 70:3–15Google Scholar
  9. 9.
    Kahn MF, Khan MA (1994) The SAPHO syndrome. Baillieres Clin Rheum 8: 333–362Google Scholar
  10. 10.
    Kotilainen PM, Laxen FO, Manner IK, Gullichsen RE, Saario RM (1996) An aseptic inflammation of the clavicle in a patient with Crohn's disease — a potential manifestation of the SAPHO syndrome. Scand J Rheumatol 25:112–114Google Scholar
  11. 11.
    Trotta F, La Corte R, Bajocchi G, Manicardi S (1990) Hyperostosis and multifocal osteitis: a purely rheumatological subset of the SAPHO syndrome. Clin Exp Rheumatol 8:401–405Google Scholar
  12. 12.
    Ueno K, Rikimaru S, Kawashima Y, Sakai H (1986) Bone imaging of sternocostoclavicular hyperostosis in palmoplantar pustulosis. Clin Nucl Med 11:420–425Google Scholar
  13. 13.
    Vecchio PC, Marlton PV, Webb J (1992) Noninfectious osteitis: part of the SAPHO syndrome. Clin Rheumatol 11:371–375Google Scholar

Copyright information

© Springer-Verlag 1997

Authors and Affiliations

  • P. Kotilainen
    • 1
  • R. E. Gullichsen
    • 2
  • R. Saario
    • 1
  • I. Manner
    • 3
  • E. Kotilainen
    • 4
  1. 1.Department of Medicine, University of TurkuTurku University Central HospitalTurkuFinland
  2. 2.Department of SurgeryUniversity of TurkuTurkuFinland
  3. 3.Department of Diagnostic RadiologyUniversity of TurkuTurkuFinland
  4. 4.Department of Surgery and NeurosurgeryUniversity of TurkuTurkuFinland

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