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Rheumatology Advance Access originally published online on June 16, 2009
Rheumatology 2009 48(8):968-971; doi:10.1093/rheumatology/kep157
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© The Author 2009. Published by Oxford University Press on behalf of the British Society for Rheumatology. All rights reserved. For Permissions, please email: journals.permissions@oxfordjournals.org

Rituximab treatment of the anti-synthetase syndrome—a retrospective case series

Marthe Sem1,2,*, Øyvind Molberg1,*, May Brit Lund3 and Jan Tore Gran1

1Department of Rheumatology, Rikshospitalet University Hospital, 2Faculty of Medicine, University of Oslo and 3Department of Respiratory Medicine, Rikshospitalet University Hospital, Oslo, Norway.

Correspondence to: Øyvind Molberg, Department of Rheumatology, Rikshospitalet University Hospital, N-0027 Oslo, Norway. E-mail: oyvind.molberg{at}medisin.uio.no


   Abstract

Objective. Interstitial lung disease (ILD) is the major determinant of morbidity and mortality in the anti-synthetase syndrome (ASS). Here we have retrospectively assessed 11 ASS patients with ILD treated with the anti-CD20 mAB rituximab at our tertiary referral hospital.

Methods. Data on clinical and laboratory parameters, lung imaging by high-resolution CT thorax and pulmonary function tests were collected from patient examinations done up to 6 months before rituximab was initiated, and at 3 and 6 months post-treatment.

Results. All the 11 ASS patients had severe and progressive ILD and most of them had previously failed on cyclophosphamide and/or other immuno-modulating agents. Rituximab appeared to stabilize and/or improve the ILD in 7 of 11 ASS patients during the first 6 months after treatment. The rituximab treatment appeared to decrease the serum level of anti-Jo-1 antibodies, but the decrease was most often modest. One patient developed a fatal infection 3 months after the last infusion with rituximab. In the other ASS patients, the treatment was well tolerated.

Conclusions. This retrospective case series indicates a short-term beneficial effect of rituximab in ASS. Prospective, controlled studies are needed to validate this finding and further assess safety issues.

KEY WORDS: Rituximab, Anti-synthetase syndrome, Treatment, Anti-Jo-1, Anti-aminoacyl tRNA synthetase


*Marthe Sem and Øyvind Molberg equally contributed to this work.

Submitted 20 February 2009; revised version accepted 14 May 2009.
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