Refractory multicentric reticulohistiocytosis treated by infliximab: two cases

J. Sellam¹, C. Job Deslandre¹, F. Dubreuil², S. Arfi², A. Kahan¹

¹René Descartes University, Rheumatology A Department, Cochin Hospital, AP-HP, Paris, France; ²Rheumatology and Internal Medicine Department, CHU de Fort-De-France, Martinique.

ABSTRACT
We report the effect of infliximab, a monoclonal anti-TNFa antibody, in two patients with refractory cutaneous and articular multicentric reticulohistiocytosis (MRH).
One 37-year-old woman and one 53-year-old woman with polyarthritis, facial rash and nodular lesions on the hands related to MRH were refractory to multiple agents: cariolysine, corticosteroids, hydroxychloroquine and cytotoxic agents. Infliximab at 3 mg/kg which was then increased to 5 mg/kg in combination with methotrexate or azathioprine was effective on cutaneous manifestations of the disease but not on polyarthritis. A switch to etanercept did not improve polyarthritis in the second patient.
Some data suggest that TNFa is involved in MRH, but based on our cases anti-TNFa therapy needs further evaluation in patients with refractory MRH.

Key words
Multicentric reticulohistiocytosis, infliximab, anti-TNFa, monoclonal antibody.

Please address correspondence to: Dr. Chantal Job Deslandre, Cochin Hospital, Rheumatology A Department, AP-HP, 27 Rue du Faubourg St-Jacques, 75014 Paris, France.
E-mail: chantal.deslandre@cch.ap-hop-paris.fr

Clin Exp Rheumatol 2005; 23: 97-99.
© CLINICAL AND EXPERIMENTAL RHEUMATOLOGY 2005.