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.
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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
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
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
Cabay JE, Marcelis, Dondelinger RF (1998) La spondylodiscite inflammatoire comme manifestation radiologique unique du SAPHO. J Radiol 79:337–340
Cotten A, Flipo RM, Mentre A, Delaporte E, Dusquesnoy B, Chastenet P (1995) SAPHO syndrome. Radiographics 15:1147–1154
Davies AM, Marino AJ, Evans N, Grimer RJ, Deshmukh N, Mangham DC (1999) SAPHO syndrome: 20-year follow-up. Skeletal Radiol 28:159–162
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
Earwaker JW, Cotten A (2003) SAPHO: syndrome or concept? Imaging findings. Skeletal Radiol 32:311–327
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
Kahn MF, Chamot AM (1992) SAPHO syndrome. Rheum Dis Clin North Am 18:225–246
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
Marshall H, Bromilow J, Thomas AL, Arden NK (2002) Pamidronate: a novel treatment for the SAPHO syndrome? Rheumatology 41:231–233
Nachtigal A, Cardinal E, Bureau NJ, Sainte-Marie LG, Milette F (1999) Vertebral involvement in SAPHO syndrome: MRI findings. Skeletal Radiol 28:163–168
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
Toussirot E, Dupond JL, Wending D (1997) Spondylodiscitis in SAPHO syndrome. A series of eight cases. Ann Rheum Dis 56:52–58
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
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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
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DOI: https://doi.org/10.1007/s00586-004-0791-4