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Rheumatology Advance Access originally published online on May 20, 2008
Rheumatology 2008 47(7):1051-1053; doi:10.1093/rheumatology/ken172
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© The Author 2008. Published by Oxford University Press on behalf of the British Society for Rheumatology. All rights reserved. For Permissions, please email: journals.permissions@oxfordjournals.org

Interferon-{alpha}—a new therapeutic option in refractory juvenile Behçet's disease with CNS involvement

J. B. Kuemmerle-Deschner1, N. Tzaribachev1, C. Deuter2, M. Zierhut2, M. Batra3 and I. Koetter4

1Division of Paediatric Rheumatology, University Children's Hospital Tuebingen, 2Centre for Ophthalmology, 3Department of Neuroradiology and 4Department of Internal Medicine, Division of Haematology, Oncology, Immunology, Rheumatology and Pulmology, University Hospital Tuebingen, Tuebingen, Germany.

Correspondence to: J. B. Kuemmerle-Deschner, Division of Pediatric Rheumatology, University Children's Hospital Tuebingen, Hoppe-Seyler-Strasse 1, 72076 Tübingen, Germany. E-mail: kuemmerle.deschner{at}uni-tuebingen.de


  Abstract

Objective. To report the successful treatment with recombinant human IFN- {alpha} 2a (rhIFN-{alpha}2a) in two male adolescents suffering from severe treatment-resistant Behçet's disease (BD) with central nervous system (CNS) involvement.

Methods. The patients were 14- and 15-yrs old. Both met the International Study Group for Behçet's disease, O’Duffy and the Japanese criteria for the classification or diagnosis of BD. Signs of CNS involvement were impaired sensorimotor function of the left arm, hemiparesis of right arm and leg, dizziness and walking instability in Patient 1, weakness of both legs, impaired bladder-, bowel- and sexual function in Patient 2 and vasculitic lesions on cranial MRI in both patients. RhIFN-{alpha}2a was administered initially at 3 million IU/day for 4 weeks followed by 3 x 3 million IU/week.

Results. Complete remission was achieved in Patient 1 (reduction in BD activity score from 17 to 2). Patient 2 experienced remarkable improvement (reduction of BD activity score from 23 to 15). In both patients the MRI lesions improved. Patient 2 had mild flu-like symptoms as adverse effect.

Conclusion. RhIFN-{alpha}2a was effective and well tolerated in these juvenile patients with severe neurological BD. Regarding the serious consequences following ocular and CNS affection and adverse effects of steroid dependency, administration of rhIFN-{alpha}2a at an earlier time point needs to be considered.

KEY WORDS: Behçet's disease, Juvenile, Central nervous system, Interferon-{alpha}

Submitted 6 August 2007; revised version accepted 2 April 2008.
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