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Rheumatology (Oxford). 2010 Aug;49(8):1505-12. doi: 10.1093/rheumatology/keq122. Epub 2010 Apr 29.

Biologic therapy in refractory chronic non-bacterial osteomyelitis of childhood.

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  • 1Department of Rheumatology, Institute of Child Health and Great Ormond Street Hospital, London WC1N 1EH, UK. d.eleftheriou@ich.ucl.ac.uk

Abstract

OBJECTIVE:

To date there is no uniformly effective treatment for either chronic recurrent multifocal osteomyelitis (CRMO) or synovitis, acne, pustulosis, hyperostosis and osteitis (SAPHO) syndrome. We report on our clinical experience of using biologic therapy to treat children with these conditions.

METHODS:

Retrospective descriptive case series of four children with refractory disease treated with biologics. Disease activity was assessed at predetermined time points (T = 0, T = 6 weeks and T = 12 months after the start of biologic therapy, and at latest follow-up) using a combination of clinical examination and radiological findings: a 10 cm pain and physician visual analogue scale; the Childhood Health Assessment Questionnaire as an assessment of disability; and changes in markers of systemic inflammation.

RESULTS:

There was an initial improvement in all parameters assessed for all three children treated with TNF-alpha blockade, although the third case had to discontinue the therapy due to a suspected (but unconfirmed) fungal skin infection. Anakinra treatment alleviated the symptoms in the fourth patient at 6 weeks, but there was no sustained response to treatment at 1-year follow-up.

CONCLUSION:

We present our preliminary experience of using biological therapies to treat children with CRMO and SAPHO in conjunction with other immunosuppression. Further studies are needed to establish the role of these therapies in refractory CRMO and SAPHO.

PMID:
20430869
DOI:
10.1093/rheumatology/keq122
[PubMed - indexed for MEDLINE]
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