184 research outputs found
Tumour necrosis factor alpha blockade in treatment resistant pigmented villonodular synovitis
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 ((90)Y) failed to
control the disease. After finding marked tumour necrosis factor alpha
(TNF alpha) expression in arthroscopic synovial tissue samples, treatment
with an anti-TNF alpha 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 TNF alpha
expression in the synovium. DISCUSSION: This is probably the first case
which describes treatment with TNF alpha 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 TNFalpha
blockade treatment in refractory PVNS
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