Document Detail

Multicentric reticulohistiocytosis associated arthritis responding to anti-TNF and methotrexate.
MedLine Citation:
PMID:  21485770     Owner:  NLM     Status:  MEDLINE    
We present a patient with therapy resistant multicentric reticulohistiocytosis (MRH). MRH is a rare granulomatous, multisystem disease characterised most frequently by 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 TNFalpha 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 TNFa 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.
K J De Knop; N E Aerts; D G Ebo; J F Van Offel; W J Stevens; L S De Clerck
Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Acta clinica Belgica     Volume:  66     ISSN:  1784-3286     ISO Abbreviation:  Acta Clin Belg     Publication Date:    2011 Jan-Feb
Date Detail:
Created Date:  2011-04-13     Completed Date:  2011-05-24     Revised Date:  2014-03-18    
Medline Journal Info:
Nlm Unique ID:  0370306     Medline TA:  Acta Clin Belg     Country:  Belgium    
Other Details:
Languages:  eng     Pagination:  66-9     Citation Subset:  IM    
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MeSH Terms
Anti-Inflammatory Agents / therapeutic use*
Antibodies, Monoclonal / therapeutic use*
Arthritis / drug therapy*,  epidemiology*
Hand / radiography
Histiocytosis / drug therapy*,  etiology*,  pathology
Immunosuppressive Agents / therapeutic use*
Lupus Erythematosus, Systemic / complications*
Methotrexate / therapeutic use*
Middle Aged
Synovitis / drug therapy,  ultrasonography
Tumor Necrosis Factor-alpha
Reg. No./Substance:
0/Anti-Inflammatory Agents; 0/Antibodies, Monoclonal; 0/Immunosuppressive Agents; 0/Tumor Necrosis Factor-alpha; 0/infliximab; YL5FZ2Y5U1/Methotrexate

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