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Rheumatology Advance Access originally published online on April 9, 2008
Rheumatology 2008 47(6):877-880; doi:10.1093/rheumatology/ken074
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© 2008 The Author(s)
This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/2.0/uk/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.


Effectiveness of infliximab in the treatment of refractory juvenile dermatomyositis with calcinosis

P. Riley1, L. J. McCann1, S. M. Maillard1, P. Woo1, K. J. Murray2 and C. A. Pilkington1

1Juvenile Dermatomyositis Research Centre, University College London, Institute of Child Health, London, UK and 2Rheumatology Department, Princess Margaret Hospital for Children, Perth, Western Australia, Australia.

Correspondence to: C. A. Pilkington, Juvenile Dermatomyositis Research Centre, Institute of Child Health UCL, 30 Guilford Street, London WC1N 1EH, UK. E-mail: c.pilkington{at}ich.ucl.ac.uk


   Abstract

Objectives. Some juvenile dermatomyositis (JDM) patients have a disease course which is refractory to multiple drug treatments. Prolonged disease activity is associated with increased mortality and morbidity. TNF-{alpha} has been identified in high levels in JDM patients who have a long disease course and calcinosis. We assessed the response of five refractory JDM patients to the anti-TNF-{alpha} monoclonal antibody, infliximab.

Methods. For all five patients intravenous infliximab was initially given at a dose of 3 mg/kg. Further doses were then given at weeks 2, 6 and every 8 weeks thereafter. The dose and frequency were tailored in accordance with clinical response. Clinical and laboratory data were collected prospectively.

Results. We report results between 8 and 30 months after starting infliximab. Improvements were seen in all five patients as shown by positive changes in physician visual analogue scale (VAS), Childhood Myositis Assessment Score (CMAS), Childhood Health Assessment Questionnaire (CHAQ), joint range of movement and, in some, regression of calcinosis and skin signs. There were no major side effects observed with addition of infliximab to the therapeutic regime.

Conclusions. Major clinical benefit was demonstrated after the initiation of infliximab in all five cases of refractory JDM.

KEY WORDS: Juvenile dermatomyositis, Calcinosis, Refractory, Anti-tumour necrosis factor treatment, Infliximab

Submitted 30 March 2007; revised version accepted 31 January 2008.
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