Refractory multicentric reticulohistiocytosis treated by infliximab: two cases
Sellam, J.; Deslandre, C.J.; Dubreuil, F.; Arfi, S.; Kahan, A.
Clinical and Experimental Rheumatology 23(1): 97-99
2005
ISSN/ISBN: 0392-856X PMID: 15789895 Document Number: 590842
We report the effect of infliximab, a monoclonal anti-TNFalpha 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 TNFalpha is involved in MRH, but based on our cases anti-TNFalpha therapy needs further evaluation in patients with refractory MRH.