Case reportTwo cases of Takayasu's arteritis occurring under anti-TNF therapy
Introduction
Takayasu arteritis is a rare, systemic large-vessel vasculitis of unknown aetiology that most commonly affects women of childbearing age. It is a granulomatous inflammatory disease that involves large- and medium-size arteries, with a predilection for the aorta and its branches. The association of Takayasu's arteritis and rheumatic diseases, including rheumatoid arthritis [1] and ankylosing spondylitis [2], [3], [4] has been described by several authors.
Tumor necrosis factor alpha (TNFa) is a cytokine involved in systemic inflammation, produced primarily by macrophages, but also by a broad variety of cell types including lymphoid cells, mast cells, endothelial cells; it promotes macrophage activation and lymphocyte trafficking and homeostasis. Since TNFa is implicated in other granulomatous diseases, its role in the pathogenesis of Takayasu's arteritis has been postulated and at least two large studies [5], [6] and several case reports [7], [8], [9] documented the efficacy of TNFa inhibitors (in particular the chimeric monoclonal antibody infliximab and the fusion protein etanercept) in cases of Takayasu's arteritis resistant to conventional glucocorticoid treatment.
In the present report, we describe two cases of Takayasu's arteritis in two patients with rheumatic diseases under treatment with TNFa inhibitors (rheumatoid arthritis treated with adalimumab and HLA-B27 negative spondylarthropathy treated with golimumab).
Section snippets
Case 1
A 43-year-old South American female patient formerly diagnosed with seropositive erosive rheumatoid arthritis in remission under treatment with adalimumab (40 mg biweekly since 5 years) and methotrexate (MTX, 25 mg weekly) was referred to our clinic with suspicion of Takayasu's arteritis. A thoraco-abdominal computerised tomography (CT-scan) was performed as part of a diagnostic investigation into persistently raised acute phase response in the absence of clinical signs or symptoms of active
Case 2
A 32-year-old female patient of Srilankan origin, diagnosed with a HLA-B27 negative spondylarthropathy with axial and peripheral manifestations, presented with a 3-week history of left arm claudication with weakness and local hypothermia.
Her recent clinical history was relevant only for type II diabetes treated with glicazide. Her spondylarthropathy (with axial and peripheral involvement) was treated with golimumab that was started recently (50 mg monthly, two doses) with a good clinical
Discussion
The treatment of relapsing or resistant Takayasu's arteritis with anti TNFa therapy has been proven effective in several case reports and two larger studies [5], [6], [10], [11]. However, autoimmune phenomena arising while patients are undergoing TNFa inhibition are all but rare and vasculitis, together with drug induced lupus, are the most common systemic manifestations [12], [13]. In most cases, vasculitis presents as a cutaneous manifestation, but renal, peripheral nerve system and lung
Conclusion
Our report is the first one to show the development of Takayasu's arteritis under treatment with TNFa inhibitors in patients with rheumatic diseases; retrospective analysis of large cohorts of patients under this drug class could help us to confirm a causative link and define subsets of patients susceptible of developing the disease.
Disclosure of interest
The authors declare that they have no conflicts of interest concerning this article.
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