Skip to main content

Advertisement

SpringerLink
Log in

Isolated thoracic spine lesion: is this the presentation of a SAPHO syndrome? A case report

  • Ideas and Technical Innovations
  • Published:
European Spine Journal Aims and scope Submit manuscript

Abstract

A case of an isolated lesion of the thoracic spine attributed to SAPHO syndrome is presented. A 51-year-old man was referred for inflammatory pain in the thoracic spine. The general examination was normal (especially cutaneous and rheumatologic examinations). Laboratory analysis showed only a mild inflammatory reaction. Standard radiographs showed partial condensation of T8. Computed tomography showed osteolysis of the anterior corner of T8, and MRI revealed an abnormal signal of T8, with enlargement of the prevertebral soft tissue. Percutaneous and thoracoscopic biopsies showed a nonspecific inflammatory process, and cultures were sterile. Initially, several diagnoses were advanced: infectious spondylitis, malignant tumor, lymphomas, Paget disease, seronegative spondyloarthropathies and finally atypical SAPHO syndrome. Three months later, the patient experienced more pain. General examination was still normal. The radiological findings worsened, while the inflammatory blood tests were normal. A new thoracoscopic biopsy revealed a nonspecific inflammatory process. A diagnosis of SAPHO syndrome was made, despite the lack of typical lesions. Dramatically improving with anti-inflammatory therapy, the patient’s condition was favorable at 3-year follow-up. This atypical presentation of an isolated lesion in the spine makes the diagnosis of a SAPHO syndrome difficult but possible. Spine surgeons must be aware of this rare entity, to avoid misdiagnosis and unnecessary repeated surgical biopsies.

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
Fig. 5
Fig. 6
Fig. 7

Similar content being viewed by others

References

  1. Akisue T, Yamamoto T, Marui T, Hitora T, Nagira K, Nakatani T, Mihune Y, Kurosaka M (2002) Lumbar spondylodiscitis in SAPHO syndrome: multimodality imaging findings. J Rheumatol 29:1100–1101

    PubMed  Google Scholar 

  2. Benhamou C, Chamot A, Khan M (1988) Synovitis-acne-pustulosis hyperosteomyelitis syndrome (SAPHO):a new syndrome among the spondyloarthropathies? Clin Exp Rheumatol 6:109–112

    CAS  PubMed  Google Scholar 

  3. Boutin RD, Resnick D (1998) The SAPHO syndrome: an evolving concept for unifying several idiopathic disorders of bone and skin. AJR Am J Roentgenol 170:585–591

    CAS  PubMed  Google Scholar 

  4. Cabay JE, Marcelis, Dondelinger RF (1998) La spondylodiscite inflammatoire comme manifestation radiologique unique du SAPHO. J Radiol 79:337–340

    CAS  PubMed  Google Scholar 

  5. Cotten A, Flipo RM, Mentre A, Delaporte E, Dusquesnoy B, Chastenet P (1995) SAPHO syndrome. Radiographics 15:1147–1154

    CAS  PubMed  Google Scholar 

  6. Davies AM, Marino AJ, Evans N, Grimer RJ, Deshmukh N, Mangham DC (1999) SAPHO syndrome: 20-year follow-up. Skeletal Radiol 28:159–162

    Article  CAS  PubMed  Google Scholar 

  7. Dougados M, Van der Linden S, Julhin R et al (1991) The European Spondyloarthropathy Study Group preliminary criteria of the classification of Spondyloarthropathy. Arthritis Rheum 34:1218–1272

    CAS  PubMed  Google Scholar 

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

    CAS  PubMed  Google Scholar 

  9. Hayem G, Bouchaud-Chabot A, Benali K, Roux S, Palazzo E, et al (1999) SAPHO syndrome: a long term follow-up study of 120 cases. Semin Arthritis Rheum 29:159–171

    CAS  PubMed  Google Scholar 

  10. Kahn MF, Chamot AM (1992) SAPHO syndrome. Rheum Dis Clin North Am 18:225–246

    CAS  PubMed  Google Scholar 

  11. Kotilainen P, Gullischen RE, Saario R, Manner I, Kotilainen E (1997) Aseptic spondylitis as the initial manifestation of the SAPHO syndrome. Eur Spine J 6:327–329

    CAS  PubMed  Google Scholar 

  12. Marshall H, Bromilow J, Thomas AL, Arden NK (2002) Pamidronate: a novel treatment for the SAPHO syndrome? Rheumatology 41:231–233

    Article  CAS  Google Scholar 

  13. Nachtigal A, Cardinal E, Bureau NJ, Sainte-Marie LG, Milette F (1999) Vertebral involvement in SAPHO syndrome: MRI findings. Skeletal Radiol 28:163–168

    Article  CAS  PubMed  Google Scholar 

  14. Olivieri I, Padula A, Ciancio G, Salvarani C, Niccoli L, Cantini F (2002) Successful treatment of SAPHO syndrome with infliximab: report of two cases. Ann Rheum Dis 61:375–376

    Article  CAS  PubMed  Google Scholar 

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

    CAS  PubMed  Google Scholar 

  16. Wagner AD, Andresen J, Jendro MC, Hülsemann JL, Zeidler H (2002) Sustained response to tumor necrosis factor α-blocking agents in two patients with SAPHO syndrome. Arthritis Rheum 46:1965–1968

    Article  PubMed  Google Scholar 

Download references

Author information

Authors and Affiliations

  1. Department of Orthopedic and Traumatologic Surgery, University Paris-Sud Hospital of Bicêtre, Le Kremlin Bicêtre, France

    Charles Court, Céline Charlez, Véronique Molina & Jacques Yves Nordin

  2. Department of Radiology, University Paris-Sud Hospital of Bicêtre, Le Kremlin Bicêtre, France

    Anne Miquel

  3. Department of Rheumatology, University Paris-Sud, Hospital of Bicêtre, Le Kremlin Bicêtre, France

    Didier Clerc

  4. Orthopedic and Traumatology Surgery Service of Bicêtre, 78, rue du général Leclerc, 94270, Le Kremlin Bicêtre, France

    Charles Court

Authors
  1. Charles Court
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Céline Charlez
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Véronique Molina
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Didier Clerc
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Anne Miquel
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. Jacques Yves Nordin
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Charles Court.

Rights and permissions

Reprints and permissions

About this article

Cite this article

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

Download citation

  • Received:

  • Revised:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00586-004-0791-4

Keywords

Search

Navigation