Refractory multicentric reticulohistiocytosis treated by infliximab: two cases.
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.
Sellam
J
J
René Descartes University, Rheumatology A Department, Cochin Hospital, AP-HP, Paris, France.
Deslandre
C Job
CJ
Dubreuil
F
F
Arfi
S
S
Kahan
A
A
D000328:Adult; D000911:Antibodies, Monoclonal / Q000627:therapeutic use*; D018501:Antirheumatic Agents / Q000627:therapeutic use*; D005260:Female; D015616:Histiocytosis, Non-Langerhans-Cell / Q000188:drug therapy*; D006801:Humans; D000069285:Infliximab; D008875:Middle Aged; D016896:Treatment Outcome
D000911:Antibodies, Monoclonal; D018501:Antirheumatic Agents; D000069285:Infliximab