SAPHO syndrome in an adult with ulcerative colitis responsive to intravenous pamidronate: a case report and review of the literature

Abstract

International audienceSynovitis, 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

Extraskeletal benefits and risks of calcium, vitamin D and anti-osteoporosis medications

“…Several case reports have shown successful therapy with infusions of pamidronate disodium and zoledronic acid [185, 186]. …”

Clinical care of children with sterile bone inflammation

“…Corticosteroids have been reported to be successful, but side-effects limit the usefulness in long-term treatment. Perhaps the strongest data for the next step in treatment are available for the intravenous bisphophonate pamidronate [52][53][54][55][56][57][58][59][60][61][62][63][64] (see Table 1 pamidronate with resolution of bone inflammation on MRI after 6 months of treatment. Four patients had MRI confirmed recurrence of CRMO, which responded to one pamidronate retreatment.…”

Expression of Tumor Necrosis Factor-Alpha (TNF-α TNF-α Converting Enzyme and Matrix Metalloproteinase-3 in SAPHO Syndrome Synovium - A Rare Case Accompanied by Acrodermatitis Continua of Hallopeau: A Case Report and Review of Anti-TNF-α Therapy

“…In cases not respond to the first-line treatment, BPs could be a good therapeutic modality except anti-TNF-α agents. Recently some successful treatments of SAPHO syndrome with BPs have been reported (Amital et al, 2004;Colina et al, 2009;Siau & Laversuch, 2010). BPs are potent inhibitors of osteoclastic bone resorption and are an important therapeutic modality in the management of Paget's disease, multiple myeloma, malignancy-associated hypercalcaemia, bone metastasis and osteoporosis.…”