RT Journal Article
SR Electronic
T1 Tumour necrosis factor α blockade in treatment resistant pigmented villonodular synovitis
JF Annals of the Rheumatic Diseases
JO Ann Rheum Dis
FD BMJ Publishing Group Ltd and European League Against Rheumatism
SP 497
OP 499
DO 10.1136/ard.2004.025692
VO 64
IS 3
A1 E-J A Kroot
A1 M C Kraan
A1 T J M Smeets
A1 M Maas
A1 P P Tak
A1 J M G W Wouters
YR 2005
UL http://ard.bmj.com/content/64/3/497.abstract
AB Background: Pigmented villonodular synovitis (PVNS) is considered to be a neoplastic-like disorder of the synovium histologically characterised by villonodular hyperplasia, resulting in dense fibrosis and haemosiderin deposition. The pathogenesis of the disease is still unknown. Case report: A patient presented with severe treatment resistant PVNS of the right knee joint. Several conventional treatment regimens, including open surgical synovectomy and intra-articular injections of yttrium-90 (90Y) failed to control the disease. After finding marked tumour necrosis factor α (TNFα) expression in arthroscopic synovial tissue samples, treatment with an anti-TNFα monoclonal antibody (infliximab) at a dose of 5 mg/kg was started. Additional courses with the same dose given 2, 6, 14, and 20 weeks later, and bimonthly thereafter up to 54 weeks, controlled the signs and symptoms. Immunohistological analysis at follow up identified a marked reduction in macrophage numbers and TNFα expression in the synovium. Discussion: This is probably the first case which describes treatment with TNFα blockade of PVNS in a patient who is refractory to conventional treatment. It provides the rationale for larger controlled studies to elucidate further the efficacy of TNFα blockade treatment in refractory PVNS.