Title
Multicentric reticulohistiocytosis associated arthritis responding to anti-TNF and methotrexate Multicentric reticulohistiocytosis associated arthritis responding to anti-TNF and methotrexate
Author
Faculty/Department
Faculty of Medicine and Health Sciences
Publication type
article
Publication
Leuven ,
Subject
Human medicine
Source (journal)
Acta clinica Belgica. - Leuven, 1946 - 1997
Volume/pages
66(2011) :1 , p. 66-69
ISSN
0001-5512
ISI
000288845700015
Carrier
E
Target language
English (eng)
Full text (Publishers DOI)
Affiliation
University of Antwerp
Abstract
We present a patient with therapy resistant multicentric reticulohistiocytosis (MRH). MRH is a rare granulomatous, multisystem disease characterised most frequentlyby disfiguring papulonodular skin lesions and sometimes a destructive polyarthritis, though any organ can be involved. Abnormal histiocytic reactions to an undetermined stimulus (possibly an associated mycobacterial infection, auto immune process or neoplastic process) have been proposed as an underlying mechanism. The diagnosis is confirmed by histopathology of the cutaneous nodules and/or synovial membrane by the presence of CD68-positive histiocytes and multinucleated giant cells with an eosinophilic ground-glass cytoplasm. Recent studies have identified TNFα and other inflammatory cytokines to be highly expressed in the synovium and synovial fluid of affected joints in patients with MRH. Based on these findings, we treated our patient with infliximab in combination with methotrexate with marked improvement of morning stiffness, tender and swollen joint count, visual analogue scale and health assessment questionnaire after his third infusion. However, the nodules did not markedly resolve. When treating patients with MRH with TNFα neutralizing drugs, one has to keep the possible association with malignancy in 15-30% of cases in mind and these products should be used with caution.
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