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Abstract Number: 2968

Efficacy of Anti-TNF Alpha in Severe and Refractory Cardiovascular Involvement  of Behcet’s Disease: A Multicenter Observational Study of 18 Patients

Anne-Claire Desbois1, Olga Addimanda2, Marc Lambert3, Eric Hachulla3, F. Ackermann4, Benjamin subran5, Arnaud Hot6, Francois Maurier7, christelle mausservey8, Fanny Bernard9, tristan mirault10, Fleur Cohen11, Laurent Perard12, Gaelle Leroux13, nicolas champtiaux14, Patrice Cacoub15 and David Saadoun16, 1Hôpital Pitié-Salpêtrière, Internal Medicine and Clinical Immunology, Paris, France, 2Rheumatology Unit, Istituto Ortopedico Rizzoli, Bologna, Italy, 3CHU Lille, Département de Médecine Interne et Immunologie Clinique, F-59000 Lille, France, Lille, France, 4internal medicine, Hopital Foch, Suresnes, France, 5hopital foch, suresnes, France, 6Internal Medicine, Hopital Edouard Herriot, Lyon, France, 7Department of Internal Medicine, HP Metz Belle Isle Hospital, Metz, France, 8CH Chalon sur saone, Chalon sur saone, France, 9CHU Marseille, marseille, France, 10HEGP, paris, France, 11Internal Medicine Dpt 2, Pitié-Salpêtrière Hospital, APHP, Paris, France, 12Hôpital Edouard Herriot, Lyon, France, 13Department of Internal Medicine and Clinical Immunology, Hospital University Department: inflammation, immunopathology and biotherapy (DHU i2B), DHU 2iB Internal Medicine Referal Center for Autoimmune diseases Pitie Hospital, Paris, France, 14Pitié SAlpétrière, paris, France, 15Department of Internal Medicine, Pitié-Salpêtrière Hospital, Paris, France, 16Department of Internal Medicine and clinical Immunology. French National Reference Center for Autoimmune Diseases. DHU I2B (Inflammation, Immunotherapy and Biotherapy), UPMC, Paris VI, Hôpital Pitié Salpétrière, AP-HP, UPMC, Univ Paris 06, Paris, France

Meeting: 2016 ACR/ARHP Annual Meeting

Date of first publication: September 28, 2016

Keywords: Behcet's syndrome and treatment

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Session Information

Date: Tuesday, November 15, 2016

Session Title: Vasculitis - Poster III: Rarer Vasculitides

Session Type: ACR Poster Session C

Session Time: 9:00AM-11:00AM

Background/Purpose: In Behçet’s disease (BD), vascular complications affect more than 30% patients. Cardiovascular involvement is the main cause of death, especially for pulmonary or aortic aneurysms and large vein thrombosis. Immunosuppressants (azathioprine, methotrexate, cyclophosphamide) are not always effective and are often associated with significant side effects. TNF antagonists have been shown to be very quickly effective in severe ocular manifestations of BD. Data on their efficacy in BD vascular complications are scarce. Objectives: To evaluate the efficacy and safety of anti-TNF alpha in severe and/or refractory vascular involvements in BD.

Methods: A multicenter and observational study evaluating 18 patients with severe BD associated- cardiac or vascular disease (defined by international standards) treated with anti-TNF [infliximab (n=15) and adalimumab (n=3)]. Anti-TNF alpha antibodies were initiated because of severe aneurysmal arterial disease [lung (n=4) or the aorta/one of its branches (n=4) or peripheral (n=1)] and/or large vein thrombosis [pulmonary artery (n=7), thrombosis of the inferior vena cava (n = 4) or Budd-Chiari syndrome (n=3)] and/or cardiac involvement [aortic valve disease (n=1), left ventricular aneurysm (n=1), myocardial ischemia (n=1) or intra-cardiac thrombosis (n=2)]. Clinical remission was defined by resolution of clinical and biological symptoms and the absence of occurrence of new vascular lesions or worsening of existing vascular lesions.

Results: Eighteen patients [(89% male) with mean age of 30 [10; 55] years] were included. Fourteen (78%) were refractory to immunosuppressants (n=12) and / or high doses of systemic corticosteroids (n=2). Fifteen patients received infliximab injections [5mg / kg (n=13) and 3mg / kg (n=2)] and 3 adalimumab [40mg / 15 days]. Fourteen patients also received immunosuppressants [azathioprine (n=7), methotrexate (n=5), mycophenolate mofetil (n=2)] and 17 corticosteroids associated with anti-TNF alpha. Vascular lesions observed (before or at the same time of anti-TNF initiation) included: arterial aneurysms (n=11), [pulmonary (n=7), aortic (n=3), spleen (n=1) or peripheral (n = 3)], arterial occlusions (n=3) and / or venous thrombosis [pulmonary embolism (n=9), lower vena cava thrombosis (n=7), deep venous thrombosis of lower limbs (n = 2) or Budd-Chiari syndrome (n=4)]. Eight patients also had cardiac involvement [intracardiac thrombosis (n=4), valvulopathy (n=2), myocardial ischemia (n=2) or pericardial effusion (n=1)]. Vascular remission was achieved in 16 patients (89%), partial (n=3) or complete (n=13). No patients died and 2 relapsed under TNF-antagonists. Two patients who stopped anti-TNFα antibodies [side effect (n=1) and poor compliance (n=1)] had neurological or vascular flare 3 and 5 months after treatment cessation, respectively. Side effects were observed in 4 patients [pulmonary edema (n=1) infection (n=3)], requiring discontinuation in 2 patients. After a median follow up of 15 [4, 164] months, 16/18 were still under anti-TNF [cessation for side effects (n=2)]. The median dose of corticosteroids was significantly decreased in all patients at 12 months after treatment initiation [30 mg initially versus 6 mg, p=0.004]. At the end of follow-up, 94% achieved a dose of ≤10mg corticosteroids.

Conclusion: Anti TNFα antibodies represent an effective and safe treatment in 89% of BD patients with severe and refractory cardiovascular involvement. Results: Conclusion:


Disclosure: A. C. Desbois, None; O. Addimanda, None; M. Lambert, None; E. Hachulla, None; F. Ackermann, None; B. subran, None; A. Hot, None; F. Maurier, Novartis Pharmaceutical Corporation, 3; C. mausservey, None; F. Bernard, None; T. mirault, None; F. Cohen, None; L. Perard, None; G. Leroux, None; N. champtiaux, None; P. Cacoub, None; D. Saadoun, None.

To cite this abstract in AMA style:

Desbois AC, Addimanda O, Lambert M, Hachulla E, Ackermann F, subran B, Hot A, Maurier F, mausservey C, Bernard F, mirault T, Cohen F, Perard L, Leroux G, champtiaux N, Cacoub P, Saadoun D. Efficacy of Anti-TNF Alpha in Severe and Refractory Cardiovascular Involvement  of Behcet’s Disease: A Multicenter Observational Study of 18 Patients [abstract]. Arthritis Rheumatol. 2016; 68 (suppl 10). https://acrabstracts.org/abstract/efficacy-of-anti-tnf-alpha-in-severe-and-refractory-cardiovascular-involvement-of-behcets-disease-a-multicenter-observational-study-of-18-patients/. Accessed February 6, 2023.
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