Search

Synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome is a rare group of sterile, inflammatory osteoarticular disorders classically associated with skin lesions. It is occasionally associated with enteropathic disease such as ulcerative colitis. We present a 39-year-old patient with chronic ulcerative colitis who developed enteropathic SAPHO and responded well to pamidronate. We discuss the clinicopathological features with particular attention to bone pathology, and perform a literature review of this fascinating syndrome., (Copyright 2010. Published by Elsevier SAS.)

Four cases of chronic osteomyelitis of clavicle as primary manifestation of synovitis, acne, pustulosis, hyperostosis, osteomyelitis (SAPHO) are reported in adolescents. In all cases a typical radiographical evolution had been observed with progressive slow migration of sclerotic area from medial to lateral side of clavicle. Long-term evolution was alternation of remission and exacerbation but none of the patients healed.

Objective: To determine the clinical presentation and outcomes of chronic recurrent multifocal osteomyelitis (CRMO) in pediatric patients., Patients and Methods: Ten girls and four boys were followed up between 1993 and 1999 for CRMO diagnosed on the basis of radiographic bone lesions with, at the same sites, increased radionuclide uptake, negative microbiological specimens, and histological evidence of nonspecific osteomyelitis., Results: Mean age at CRMO was 9.6+/-3.4 years, mean disease duration was 5.3+/-2.5 years, and mean number of flares per patient was 5.9+/-3.7. Thirty-four percent of lesions were in the metaphyses of the lower limb bones, 14% in the pelvis, and 13% in the chest wall (with clavicular lesions in four patients). Three patients had skin lesions (psoriasis in two and palmoplantar pustulosis in one). Eight patients received antibiotic therapy, for 2 months at the most, to no advantage in the short term. Nonsteroidal anti-inflammatory drugs were used in all 14 patients and glucocorticoid therapy in four. Sulfasalazine was used in five patients, to good effect in four. Mean follow-up was 5.3+/-2.5 years. At last follow-up, eight patients had active disease, including one with synovitis and one with Takayashu's disease., Conclusion: As compared to SAPHO syndrome, skin lesions and chest wall involvement are less common in CRMO. The long-term prognosis is guarded: in our study only six of 14 patients were in remission at last follow-up.

Subclavian and superior vena cava obstruction complicating SAPHO (Synovitis, Acne, Pustulosis, Hyperostosis, Osteitis) syndrome has been described. We report the first case to our knowledge of iliac vein thrombosis complicating lumbar vertebral osteitis due to SAPHO syndrome. Lumbar MRI demonstrated a large tissue mass anterior to the involved vertebras and surrounding the right iliac vein. Histology of the mass showed aseptic inflammation.